A resiliency perspective of the lived experience of parenting infants and young children with cystic
Transcript of A resiliency perspective of the lived experience of parenting infants and young children with cystic
Edith Cowan University Edith Cowan University
Research Online Research Online
Theses: Doctorates and Masters Theses
2016
A resiliency perspective of the lived experience of parenting A resiliency perspective of the lived experience of parenting
infants and young children with cystic fibrosis in the context of infants and young children with cystic fibrosis in the context of
early lung disease surveillance early lung disease surveillance
Cindy Branch-Smith Edith Cowan University
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Recommended Citation Recommended Citation Branch-Smith, C. (2016). A resiliency perspective of the lived experience of parenting infants and young children with cystic fibrosis in the context of early lung disease surveillance. https://ro.ecu.edu.au/theses/1805
This Thesis is posted at Research Online. https://ro.ecu.edu.au/theses/1805
A Resiliency Perspective of the Lived Experience of Parenting Infants and Young Children with Cystic
Fibrosis in the Context of Early Lung Disease Surveillance
Cindy A Branch-Smith
This thesis is submitted in fulfilment of the requirements for the degree of
Doctor of Philosophy in Psychology
Edith Cowan University
School of Arts and Humanities
Date of Submission: 31st July 2016
Edith Cowan University
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USE OF THESIS
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ii Parenting Children with Cystic Fibrosis
Preface
The linking of hope and empowerment can be seen in a description of the virtues of hope in
the book ‘The Anatomy of Hope’ by Jerome Groopman (2005) who stated “hope can arrive only
when you realise that there are real options and that you have genuine choices. Hope can flourish
only when you believe that what you do can make a real difference, that your actions can bring a
future different from the present. To have hope, then, is to acquire a belief in your ability to have
some control over your circumstances. You are no longer entirely at the mercy of forces outside
yourself” (p. 26).
For parents dealing with their young child’s chronic disease, existence of hope for their child,
for future generations inflicted with the condition and hope for a cure can be generated through
advancements in science and technology, and clinical agendas that allow early tracking of the course
of disease progression. A Western Australian mother of a 4 year old child living with cystic fibrosis
commented on how she felt about being able to track her child’s condition through cutting-edge
early surveillance techniques. The linking of hope and empowerment described in Jerome
Groopman’s book is woven through her statement - I just think that you've got somewhere to start.
If you've got nowhere to start then you can’t do anything, so for me this is a starting point. I've got
information and I know what the state of her lungs are like. She's 4 and I hope she has a long life and
I hope we can keep her lungs as healthy as we can, and this is where we start with it. So as I said, I'm
grateful that it's there, I'm grateful that I can participate in it. Firstly to get information for ourselves
but secondly to help with research to help others and, ultimately to find a cure. There's lots to learn
about CF I've learnt.
Emulating transition of writing styles here in the preface, the reader will discern a change
from an academic style to a more conversational tone from chapter 3 onwards from the academic
setting where the literature on parenting and CF is situated, into my data collection methods and the
world of my informants. That is, I’m choosing to stay true to my informants’ existential experience
and not to dress it with psychological terminology.
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Abstract
Scientific and technological advances over the past few decades have contributed to an exponential
increase in life expectancy for infants born with cystic fibrosis (CF), which can cause fatal lung
disease. There has been a paradigmatic shift from reactive treatment towards early disease
detection and aggressive intervention of paediatric CF. Previous research has investigated parents’
experiences of the diagnosis phase and later life stages such as adolescence and transition to
adulthood. Less is understood about the experience of parenting infants and young children with CF,
and no research addresses this during early surveillance for CF lung disease. Early surveillance is
rapidly emerging as the likely framework for future therapeutic intervention trials in young children,
nationally and internationally. Thus, understanding parents’ mental health is essential to providing
support to families during their child’s intensive, early treatment.
This research aimed to explore how parents construct their lived experiences of parenting and how
they attribute meaning to these experiences. Particular attention was given to coping strategies,
informed by a salutogenic model of protective factors that may contribute to family adaptation and
resilience. A qualitative methodology, guided by theoretical underpinnings of phenomenology and
constructivism was used to explore parents’ experiences. Semi-structured interviews were
conducted with 67 parents (46 mothers and 21 fathers aged between 23 and 52 years) across sites
where early surveillance for CF lung disease operates in Australia. A thematic analytic approach was
used to analyse the data.
Data revealed that whilst parents described adverse psychological consequences of their child’s
participation in early surveillance, it was also evident that beneficial outcomes eclipsed adversities.
Five major themes represent the emergent structure of parents’ lived experience: Redefined
Expectations and Reimagined Identity, Redefined Reality; Understanding the Unknown and
Understanding Uncertainty; Good Days and Bad Days, Fluctuation between Positive and Negative
Outlook On Life; Early Surveillance is a Significant Event; And Early Surveillance is in the Best Interest
of my Child. Collectively, these highlight that only attending to adverse experiences and outcomes
would be to overlook significant beneficial psychological experiences and outcomes of this unique
parenting experience. Pathways that enable adaptation to paediatric chronic disease in the context
of early surveillance were also identified using a resiliency framework of family adjustment and
adaption. These findings contribute to current conceptualisations of parenting children with chronic
conditions undergoing modern interventions that aim to delay disease. This research may also
inform policy-making and models of best paediatric clinical practice, particularly those embracing a
biopsychosocial model of care that promotes parents’ mental health, as well as family resilience and
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adaptation. The importance of these findings underscores the need for further research to expand
our understanding of the parenting experience in the unique context of early surveillance.
COPYRIGHT AND ACCESS DECLARATION
I certify that this thesis does not, to the best of my knowledge and belief:
(i) incorporate without acknowledgement any material previously submitted for a degree or diploma in any institution of higher education;
(ii) contain any material previously published or written by another person except where due reference is made in the text; or
(iii) contain any defamatory material.
Signed (signature not included in this version of the thesis)
Date……………………………………….
v Parenting Children with Cystic Fibrosis
Acknowledgements
While my name is alone on the front cover of this dissertation, I am by no means its sole contributor. I would like to acknowledge those who have supported me over the past 4 years (and beyond) and who, in turn, have contributed to the completion of this doctoral dissertation. Firstly, I would like to thank the parents from Princess Margaret Hospital and The Royal Children’s Hospital who volunteered their time to participate in my research. Without you this study would not have been possible. I appreciate your willingness and your trust and openness throughout the interviews. Thank you for sharing your experiences with me. To my principal supervisor, Professor Julie Ann Pooley, thank you for your support and encouragement throughout completion of my doctoral dissertation. You are much more than a PhD supervisor to me. Your guidance has inspired me to look beyond what I can see, not only in my research, but in my personal life. I would like to thank my co-supervisors, Professor Linda Shields, Doctor Tonia Douglas and Professor Stephen Stick, for their contribution to my doctoral dissertation. A special thanks for the valuable feedback and for what they have given of their time and skills. The Telethon Kids Institute has been a wonderful place to conduct my research and I look forward to a career in child and parent mental health here. I would like to especially acknowledge Doctor Clair Lee for her encouragement and support on both a professional and personal level. As a high-achieving, generous and genuine woman, you are a role model for me, and I thank you for sharing your personality and insights with me. Thank you to Liz Balding, CF clinic nurse at Princess Margaret Hospital for conceptualising much of the paediatric psychosocial research. Your never-ending endeavour to support children with CF, and their families has enabled greater understanding of what it is like to parent children living with chronic conditions. Thank you to Associate Professor John Massie of The Murdoch Children’s Research Institute for your support and advice. I thank you for giving me some of your time and encouraging me. I would also like to thank Judith Glazner of The Murdoch Children’s Research Institute for your assistance in recruitment of a national sample of participants for my research. Thank you to Cystic Fibrosis Western Australia, The Graduate Women of Western Australia and The Friends of the Institute who provided partial funding for this work. I would like to express a deep sense of gratitude to my parents, Anne and Gary Herren. Thank you for your unconditional love and for your overwhelming support of my education. Throughout this journey Mum, you have been my voice of reason, my inspiration, my role model, my shoulder to cry on, my listening ear, and my cheerleader. Thank you to AnneMarie Naylor and Anne Herren for dotting the i’s and crossing the t’s, and thank you to Lousie Branch-Smith for the DOIs! To my friend Linda, you’re support and encouragement have been a shining light in dark times. Your phone calls and city visits are always appreciated. The homestead villa stays have been a wonderful way to refocus, stay fresh and push on. You’ve had an uncanny ability to help me see beyond the trees in the forest to include the grain of the wood. You are a true friend.
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To my friend Michelle, though you probably didn’t know it, you have been a source of inspiration when times were tough and when I doubted myself. Your strength is a shining light, and so is your determination to get the most out of life. You are a treasured friend. My final mention is for Romesh Perera, who has learned who I am as I have grown throughout this process. Thank you for supporting, encouraging and loving me. I acknowledge the inherent difficulty that must go with partnering someone completing a PhD. I look forward to continuing our never-ending journey together.
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Dedication
I dedicate this doctoral dissertation to my Sulli-girl. We started this journey together and you saw me through to the end. Along the way you taught me patience, stability, and responsibility. More importantly, you taught me again how to love and how to be loved. I’ll miss you always, and I’m forever grateful that our paths crossed.
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Publications Arising From This Thesis
2015 Branch-Smith, C., Pooley, J., Shields, L., Stick, S., & Douglas, T., on behalf of AREST-CF. Early
surveillance of cystic fibrosis: Parents’ psychosocial experiences of disease knowledge and
their child’s medical procedures. Pediatric Pulmonology, 50 (S41), 432.
2014 Branch-Smith, C., Douglas, T., Shields, L., Stick, S., Massie, J., & Pooley, J., on behalf of
AREST-CF. Parental experiences of early pulmonary surveillance for children with cystic
fibrosis: A research proposal for improved family psychosocial outcomes. Working
Papers in the Health Sciences, 8 (1), 1-8.
2014 Branch-Smith, C., Pooley, J., Shields, L., Stick, S., & Douglas, T., on behalf of AREST-CF.
‘Arresting’ psychological issues for better health outcomes in parents of infants and
young children with cystic fibrosis. Journal of Cystic Fibrosis, 13, (Supp. 2), S116.
2014 Branch-Smith, C., Pooley, J., Shields, L., Stick, S., Douglas, T., & Rosenow, T., on behalf of
AREST-CF. ‘Arresting’ psychological issues for parents of infants and young children with
cystic fibrosis. Pediatric Pulmonology, 49 (S38), 438.
2013 Branch-Smith, C., Pooley, J., Shields, L., Stick, S., & Douglas, T., on behalf of AREST-CF. What
do parents experience and how do they cope with the AREST-CF early surveillance program
for infants and children with cystic fibrosis? Journal of Cystic Fibrosis, 12, (Supp. 1), S3.
ix Parenting Children with Cystic Fibrosis
Abbreviations
AREST CF: Australian Respiratory Early Surveillance Team for Cystic Fibrosis
BAL: bronchoalveolar lavage
CIHR: Canadian Institutes of Health Research
CF: cystic fibrosis
CFA: Cystic Fibrosis Australia
CFTR: cystic fibrosis transmembrane conductance regulator
CT: chest computed tomography scan
EU: European Union
GA: general anaesthetic
HCP: healthcare professional
LMA: laryngeal mask airway
NBS: newborn screening
PMH: Princess Margaret Hospital (Perth, Western Australia)
PTSS: post-traumatic stress syndrome
RCH: Royal Children’s Hospital (Melbourne, Victoria)
TIDES: The International Depression/Anxiety Epidemiological Study
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Glossary
Adaptation: the process of change by which an individual becomes better-suited to their situation or
environment (Kent, 1998).
Biopsychosocial: a theoretical model with the underlying premise that biological, psychological
(including cognitive, emotional and behavioral), and social (socio-economical, socio-environmental,
and cultural) factors, all play a significant role in human functioning in the context of disease or
illness (Santrock, 2007).
Chronic condition: a disease or illness that has a prolonged course, that does not resolve
spontaneously, for which a complete cure is rarely achieved (National Public Health Partnership,
pp.3).
Chronic sorrow: a sense of recurring, extended and profound sadness (Olshansky, 1962)
Cognitive dissonance: an emotional state occurring when two simultaneously held attitudes or
cognitions are inconsistent, or when there is conflict between belief and behaviour (Reber & Reber,
2001).
Constructivism: theoretical assumption in qualitative psychology that assumes a relativist ontology
(there are multiple realities), a subjectivist epistemology (researcher and respondent co-create
understandings), and a naturalistic (in the natural world) set of methodological procedures (Denzin
& Lincoln, 2003).
Coping: thoughts and behaviours people use to manage internal and external demands of situations
that are appraised as stressful (Lazarus and Folkman, 1984)
Covert disease progression: early disease that has advanced but goes undetected by traditional
measures.
Depression: generally, an affective state characterised by a sense of inadequacy, a feeling of
despondency, a decrease in activity or reactivity, pessimism, sadness and related symptoms (Reber
& Reber, 2001).
Diagnosis: the identification of the nature of an illness or other problem by clinical examination of
the symptoms (Kent, 1998).
Early surveillance: a set of medical procedures that allow clinicians to track the course of early, often
covert, progression of lung disease long before signs and symptoms are apparent.
Family adaptation: the outcome of a family’s efforts to bring a new level of balance and coherence,
as well as a satisfactory level of functioning, to a family crisis situation. The process to family
adaptation involves coping strategies, from an individual and family perspective (Danielson, et al.,
1993).
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Family resiliency: a family’s potential to recover and restore itself following a crisis, change, or other
stressors facing the family unit (Danielson, et al., 1993).
Hope: the feeling that what is wanted can be had or that events will turn out for the best; a desire
accompanied by expectation of or belief in fulfilment (Kent, 1998).
Intervention: The act of intervening or interceding with the intent of modifying the outcome. A
clinical intervention is usually undertaken to help treat or cure a condition (Kent, 1998).
Knowledge: facts, information, and skills acquired through experience or education (Kent, 1998).
Lived experience: Personal account of the world gained through direct, first-hand involvement in
everyday events (Kent, 1998).
Locus of control: the extent to which individuals believe they can control events affecting them
(Rotter, 1966). An internal locus of control orientation is a belief about whether the outcomes of
actions are contingent on what a person does, whereas an external locus of control orientation is a
belief that events are outside of personal control (Zimbardo, 1985).
Morbidity: the state of being diseased or unhealthy (Kent, 1998).
Mortality: the condition of one day having to die or the rate of failure or loss (Kent, 1998).
Normalisation: a cognitive and behavioural process of redefining one’s life through which ideas and
actions come to be seen as normal and become taken-for-granted, or natural, in everyday life (Kent,
1998).
Parent: any person/s defined by the family itself as the primary caregivers for the child/ren.
Paediatric: a branch of medicine dealing with the health and medical care of infants, children, and
adolescents from birth up to the age of 18 years (Kent, 1998).
Parental: intrinsic constructs associated with being a parent
Parent identity: the role and functions of parenting that give meaning of being a parent.
Parenting: behaviours associated with, or as a result of, being a parent.
Parenting competence: a system of knowledge, skills, and abilities that allow a parent to successfully
fulfil their parental responsibilities and also prevent, or deal with, crisis situations in a manner that
would contribute to their child’s development (Glăveanu, 2009).
Phenomenology: theoretical framework for qualitative research which focuses on people’s
understandings and interpretations of their experiences in their own terms, emphasising these as
explanations for their actions (Liamputtong & Ezzy, 2005). In general, phenomenological
psychological research aims to clarify situations lived by persons in everyday life (Giorgi & Giorgi,
2003).
Relinquishment: to give to another person or group (Kent, 1998).
Resiliency: an individual's ability to properly adapt to stress and adversity (Kent, 1998).
xii Parenting Children with Cystic Fibrosis
Resiliency Model of Family Stress, Adjustment and Adaptation: a theoretical model guiding the
understanding of a family’s response to a stressor and recovery from crisis (Danielson, et al., 1993).
Salutogenic: an approach focusing on factors that support human health and well-being, rather than
on factors that cause disease (Antonovsky, 1987).
Self-efficacy: A person's belief about his or her ability and capacity to accomplish a task or to deal
with the challenges of life (Bandura, 1994). Those with high self-efficacy believe in their capacity to
influences outcome of events whereas people low on self-efficacy believe they do not have capacity
to influence outcomes of events in their lives.
Therapeutic misconception: the belief that the purpose of a clinical trial is to directly benefit the
individual patient rather than to gather data to develop scientific knowledge (Appelbaum, 1982).
Treatment regimen: a systematic plan designed to improve and maintain the health of a patient
(Kent, 1998).
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Table of Contents
Use of Thesis………………………………………………………………………………………………………………………………………i
Preface .................................................................................................................................................... ii
Abstract .................................................................................................................................................. iii
Copyright and Access Declaration ........................................................... Error! Bookmark not defined.
Acknowledgements ................................................................................................................................. v
Dedication ............................................................................................................................................. vii
Publications Arising From This Thesis .................................................................................................. viii
Abbreviations ......................................................................................................................................... ix
Glossary ................................................................................................................................................... x
Table of Contents ................................................................................................................................. xiii
List of Tables ......................................................................................................................................... xix
List of Figures .........................................................................................................................................xx
List of Appendices ................................................................................................................................. xxi
CHAPTER 1: Introduction ........................................................................................................................ 1
1.1 Research context ........................................................................................................................... 2
1.1.1 The psychology of parenting in paediatric chronic disease. .................................................. 2
1.1.2 Paediatric Cystic Fibrosis in Australia ................................................................................... 10
1.1.3 Current Australian treatment management practices for infants and young children in the
21st century ................................................................................................................................... 12
1.1.4 Cystic fibrosis treatment management in infants and children from diagnosis to 7 years in
Western Australia and Victoria: Australian innovation for paediatric CF management .............. 14
1.2 The current research ................................................................................................................... 18
1.2.1 Research rationale ............................................................................................................... 18
1.2.2 Exploring the lived experience of parenting children with CF in the context of early
surveillance ................................................................................................................................... 19
1.2.2.1 Parallels drawn from medical treatment procedures……………………………………………….20
1.2.2.2 Parallels drawn from similar condition trajectories………………………….……………………..21
1.2.3 Aims of the research ............................................................................................................ 23
1.2.4 Research questions .............................................................................................................. 23
1.2.5 Implications of the research ................................................................................................ 24
1.3 Overview of the thesis ................................................................................................................ 25
xiv Parenting Children with Cystic Fibrosis
CHAPTER 2: Cystic fibrosis in the 21st century .................................................................................... 28
2.1 What is CF?.................................................................................................................................. 29
2.1.1 Prevalence, life expectancy, causes and clinical features. ................................................... 29
2.1.2 Chronology of CF into the 21st century. ............................................................................... 30
2.1.3 CF diagnosis: Current Australian practice via newborn screening. ..................................... 31
2.1.4 What is it like to be a young child with CF in the 21st century? .......................................... 32
2.1.5 Burden of early CF care on families in the 21st century. ...................................................... 34
2.1.6 Summary and conclusion. .................................................................................................... 36
2.2 Families living with CF ................................................................................................................. 36
2.2.1 Psychological adjustment to a CF diagnosis......................................................................... 36
2.2.2 Outcomes of parenting a child with CF. ............................................................................... 37
2.2.2.1 Parental stress, depression and anxiety…………………………………………………………………..37
2.2.2.2 Guilt and chronic sorrow…………………………………………………………………………………………39
2.2.2.3 Parental identity formation and parenting self-efficacy…………………………………………..41
2.2.3 Parental characteristics moderate child health outcomes. ................................................. 42
2.2.4 Summary and conclusion. .................................................................................................... 43
2.3 Coping in families with CF ........................................................................................................... 43
2.3.1 Coping with diagnosis. ......................................................................................................... 43
2.3.2 Coping with CF in the early years. ........................................................................................ 45
2.3.2.1 Hope and resiliency in the face of adversity…………………………….………………………………47
2.3.3 Summary and conclusion. .................................................................................................... 50
2.4 Aims and significance of this research ........................................................................................ 50
2.4.1 What does this study hope to achieve and why is it important? ........................................ 50
2.4.2 How can this research help families? .................................................................................. 51
2.4.3 What else can this research add to the body of knowledge? .............................................. 52
CHAPTER 3: Substantiative Theoretical and Methodological Derivatives ............................................ 54
3.1 Introduction ................................................................................................................................ 55
3.2 Theoretical frameworks .............................................................................................................. 55
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3.2.1 Transactional Model of Stress and Coping. ......................................................................... 55
3.2.2 The Resiliency Model of Family Stress, Adjustment and Adaptation. ................................. 57
3.2.3 Summary and conclusion. .................................................................................................... 62
3.3 Study design ................................................................................................................................ 63
3.3.1 Conceptualising qualitative research. .................................................................................. 63
3.3.1.1 Methodological and theoretical perspectives and implications…………………………….…66
3.3.2 Qualitative methods. ........................................................................................................... 67
3.3.2.1 Scientific rigour……………………………………………………………………………………………………….68
3.3.2.1.1 Operationalising rigour in qualitative research. ................................................................ 69
3.3.2.2 Phenomenology………………………………………………………………………………………..……….……70
3.3.2.3 Data collection technique……………………………………………………………………………………..…73
3.3.2.4 Sampling technique…………………………….…………………………………………………………………..75
3.3.2.5 Sample size and saturation………………………………………………………………………………………76
3.3.2.6 Thematic analysis……………….……………………………………………………………………………………77
3.4 Summary ..................................................................................................................................... 77
CHAPTER 4: Methods ............................................................................................................................ 79
4.1 Restatement of aims ................................................................................................................... 80
4.2 Research questions ..................................................................................................................... 80
4.3 Methodology ............................................................................................................................... 80
4.3.1 Phenomenology as theoretical framework. ........................................................................ 80
4.3.2 Constructivism as philosophical underpinnings. .................................................................. 81
4.4 Methods ...................................................................................................................................... 83
4.4.1 Sample. ................................................................................................................................. 83
4.4.2 Participant characteristics . .................................................................................................. 84
4.4.2.1 Western Australian sample……………………………………………………………………………………..85
4.4.2.2 Victorian sample……………………………………………………………………………………………………..85
4.4.3 Recruitment. ........................................................................................................................ 85
4.4.5 Ethics considerations. .......................................................................................................... 86
4.4.5.1 Research conduct………………………………………………………………………………………………….…86
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4.4.5.2 Data management…………………………………………………………………………………………………..87
4.4.5.3 Informed consent process…………………………………………………………………………………….…88
4.4.5.4 Consequences of the research………………………………………..…………………………………….…89
4.4.5.5 Role of the researcher…………………………………………………………………………….…………….…89
4.4.5.6 Additional ethical considerations………………………………………………………………………….…90
4.5 Data collection ............................................................................................................................ 90
4.5.1 Interview procedure. ........................................................................................................... 90
4.5.2 Data collection instrument. ................................................................................................. 93
4.6 Exploratory analysis .................................................................................................................... 94
4.7 Scientific rigour ........................................................................................................................... 99
4.7.1 Throughout the research process. ....................................................................................... 99
4.7.2 Data generation. ................................................................................................................ 100
4.7.3 Data coding and analysis. ................................................................................................... 101
4.7.4 Presentation of findings. .................................................................................................... 103
4.8 Summary ................................................................................................................................... 104
CHAPTER 5: Findings: The lived experience of parenting children with CF in the context of early
surveillance ......................................................................................................................................... 105
5.1 Identifying Themes.................................................................................................................... 106
5.2 Interpretation of phenomena – what is the ‘lived experience’ of parenting children with CF
within the context of early surveillance for lung disease? ............................................................. 107
5.3 Redefined expectations and reimagined identity; redefined reality ........................................ 108
5.3.1 Collapsed and redefined expectations............................................................................... 108
5.3.2 New normal. ....................................................................................................................... 112
5.4 Understanding the unknown and understanding uncertainty ................................................. 116
5.4.1 Understanding CF. .............................................................................................................. 117
5.4.2 Understanding early surveillance for CF lung disease. ...................................................... 122
5.5 Good days and bad days; Fluctuation between positive and negative outlook on life ............ 126
5.5.1 Bad days. ............................................................................................................................ 126
5.5.1.1 Fear and anxiety as outcomes of uncertainty in morbidity and mortality..…………….127
xvii Parenting Children with Cystic Fibrosis
5.5.1.2 Chronic sorrow……………………………………………………….……………………………………………..130
5.5.2 Good days. ......................................................................................................................... 132
5.5.2.1 Parenting competence supports the parental role…….………………………………………….132
5.5.2.2 Gratitude, both for their child and for early
surveillance………………….…………………………………………………….……………………………………………..135
5.5.2.3 Perceived control and power through certainty of disease severity from
early surveillance results………………………………………………….……………………………………..………..137
5.5.3 Fluctuation between good days and bad days. ................................................................. 139
5.6 Early surveillance is a significant event ..................................................................................... 140
5.6.1 Indicator of treatment efforts and an examination of parenting competency. ................ 141
5.6.2 Parental outcomes directly from early surveillance. ......................................................... 144
5.6.2.1 Lack of control and helplessness at time of relinquishment for medical
procedures………………………………………………………………..……….…………………………………………….144
5.6.2.2 Altruism for future CF generations………………………….…………………………………………….146
5.6.3 Fear from diagnosis exacerbated at annual review. .......................................................... 148
5.7 Early surveillance is in the best interest of my child ................................................................. 152
5.7.1 Perceived clinical benefit to child health – presence of the therapeutic misconception? 152
5.7.2 Cognitive dissonance results in anxiety and/or ambivalence. ........................................... 155
5.8 Construction and conceptualisation of hope ............................................................................ 160
5.9 Summary and conclusion .......................................................................................................... 163
CHAPTER 6: Parenting a child with CF in the context of early disease surveillance: Adapting to
paediatric chronic disease from a theoretical perspective ................................................................. 166
6.1 Introduction .............................................................................................................................. 167
6.2 Application of Resiliency Model of Family Stress, Adjustment and Adaptation....................... 167
6.2.1 Newly Instituted Patterns of Functioning. ......................................................................... 171
6.2.2 Situational and family appraisal. ........................................................................................ 172
6.2.2.1 Situational appraisal……………………………………………….……………………………………………..172
6.2.2.2 Family appraisal…………………………………………………………………………..…………………….….173
6.2.3 Meaning-making. ............................................................................................................... 174
6.2.4 Family resources – Strengths and coping strategies as family capability. ......................... 175
xviii Parenting Children with Cystic Fibrosis
6.2.5 Social support. .................................................................................................................... 178
6.2.5.1 Emotional support………………………………………………………………………….……………………..179
6.2.5.2 Network support…………………………………………………………………………….……………….…...179
6.2.5.3 Altruistic support…………………………………………………………………………….…………………....180
6.2.6 Coping patterns. ................................................................................................................. 180
6.1.6.1 Family resources contribute to family coping patterns………………………………….………181
6.1.6.2 Situational and family schema appraisals and meaning-making contribute to family
coping patterns………………………………………………………………….……………………………………………..182
6.2.7 The family adaptation process: Bonadaptation and maladaptation. ................................ 184
6.3 Summary and conclusion .......................................................................................................... 184
CHAPTER 7: Summary and conclusions: Key research findings, implications, strengths, limitations and
future directions ................................................................................................................................. 187
7.1 Key findings that apply to research aims and questions .......................................................... 188
7.2 The lived experience of parenting a child with CF in the context of early surveillance:
Implications for policy and practice ................................................................................................ 188
7.2.1 Policy and practice implications. ........................................................................................ 189
7.3 Parents’ constructions and attributions of meaning to early surveillance ............................... 193
7.4 Coping with parenting children with CF in the context of early surveillance: Implications for
theory, policy and practice ............................................................................................................. 195
7.4.1 Theoretical implications. .................................................................................................... 196
7.4.2 Policy and practice implications. ........................................................................................ 199
7.5 Further considerations: Future program development and implementation, and mental health
care of parents and implications for their families ......................................................................... 202
7.6 Strengths and significance of the research ............................................................................... 204
7.7 Limitations of my research........................................................................................................ 205
7.8 Directions for future research ................................................................................................... 206
7.9 Final considerations .................................................................................................................. 208
7.10 Postscript reflection ................................................................................................................ 210
References .......................................................................................................................................... 212
Appendices…………………………………………………………………………………….……………………………………………..279
xix Parenting Children with Cystic Fibrosis
List of Tables
Table 1 Group descriptions and recruitment details
Table 2 Demographic information of AREST CF parents by CF clinic location and stratified child age
Table 3 Stages of thematic analysis for qualitative psychological research
Table 4 Major themes and related sub-themes
xx Parenting Children with Cystic Fibrosis
List of Figures
________________________________________________________________________
Figure 1 The Resiliency Model of Family Stress, Adjustment and Adaptation
Figure 2 Map of Australia showing the regions where AREST CF operates
Figure 3 A conceptualisation of data management and data analysis processes
Figure 4 Illustration outlining data collection process
Figure 5 Overview of theoretical and procedural frameworks guiding the study
Figure 6 Representation of how parents psychologically reach an alternate sense of normal
family life
Figure 7 How parental psychological constructs are associated with home treatment and
parenting outcomes
Figure 8 Relationships between knowledge, assurance and control
Figure 9 How early surveillance influences perceived parenting competence and possibly self-
esteem
Figure 10 How hope is constructed by parents, and the objects of hope
Figure 11 Adaptation phase of The Resiliency Model of Family Stress, Adjustment and
Adaptation
Figure 12 Research findings applied to The Resiliency Model of Family Stress, Adjustment and
Adaptation
Figure 13 Screening and treatment of depression and anxiety: Algorithm for
parents/caregivers
xxi Parenting Children with Cystic Fibrosis
List of Appendices
__________________________________________________________________________________
Appendix A Invitational recruitment letter
Appendix B Information statement and consent form – Princess Margaret Hospital
Appendix C Information statement and consent form – Royal Children’s Hospital
Appendix D Western Australian counselling services
Appendix E Victorian counselling services
Appendix F Personal reflections
Appendix G Interview schedule
Appendix H Initial coding of interview transcripts
Appendix I Thematic mind-map - Trust
Appendix J Thematic mind-map – Cognitive dissonance
1 Parenting Children with Cystic Fibrosis
CHAPTER 1
Introduction
This chapter introduces the reader to the wider context of this research by outlining the topic of parenting a child with a chronic condition, the associated experiences and the outcomes for parents. How the parenting experience has been constructed theoretically and empirically is then briefly reviewed. Next is the introduction of paediatric cystic fibrosis and current treatment endeavours, which contextualises the purpose of the research. These discussions lead to an outline of the rationale of the research. Thus, the necessity for exploring the lived experience of parenting children with CF in context of a modern and aggressive treatment regimen is presented in conjunction with the importance of exploring parenting in context. Specifically, the two participant groups are described and the research questions detailed. The chapter concludes by discussing implications of the research before outlining the structure of the thesis.
2 Parenting Children with Cystic Fibrosis
Making the decision to have a child is momentous. It is to decide forever to have your
heart go walking around outside your body…Elizabeth Stone
1.1 Research context
1.1.1 The psychology of parenting in paediatric chronic disease.
Long-term conditions and chronic diseases are more commonly associated with older people
and less with children. However, a significant number of children live with chronic conditions
(Australian Institute of Health & Welfare, 2005). These conditions cause stress for children and their
families, and demand substantial amounts of time, energy and personal resources to cope with the
situation (Jessop & Stein 1989). Many different types of chronic diseases and conditions exist in the
paediatric population; some are present at birth and others may develop at a later stage during
infancy or childhood. While some children with chronic diseases or conditions may recover, most will
not lead normal lives and will need special care or management (Jessop & Stein 1989). However,
with appropriate management, many children can function well and live healthy lives (Australian
Institute of Health & Welfare, 2005).
In Australia, there is a lack of an agreed general definition of what constitutes chronic
disease or illness (Australian Institute of Health & Welfare, 2005). One definition from the United
States Center for Disease Control states that “a chronic disease is one that, in general terms, has a
prolonged course, that does not resolve spontaneously, and for which a complete cure is rarely
achieved” (National Public Health Partnership, pp.3). Whilst the terms chronic disease, illness, and
condition are used interchangeably in the literature, using the term condition in my thesis draws the
reader away from the biomedical aspects of disease and away from the negative connotations of
illness. Therefore, where appropriate the term chronic condition will be used in this thesis to refer to
a chronic disease or illness as defined by the United States Center for Disease Control.
In Australia, a high proportion of children are diagnosed with chronic conditions each year.
According to the Australian Bureau of Statistics National Health Surveys, 41% of children aged less
than 15 years had a long-term health condition in 2007-2008, whilst an estimated 37% had at least
one long-term condition in 2011-2012. Chronic conditions in Australian children are of primary
concern for the healthcare system and community, and families alike.
The Australian healthcare system is a major advocate for children’s health and well-being
with a number of national institutes caring for children with, and researching, paediatric diseases
and conditions such as the Children’s Medical Research Institute, Children’s Cancer Institute
Australia and The Australian Institute of Family Studies. The National Health and Medical Research
Centre and The Australian Institute of Health and Welfare are also strong advocates for children’s
health. They provide funding and guidance on a number of child-related health issues. The Telethon
Kids Institute is a local institute with a specific focus on chronic conditions in childhood. The work of
3 Parenting Children with Cystic Fibrosis
these Institutes, and the work of others nationally and internationally, has increased children’s life
expectancies for various chronic conditions exponentially over the past two to three decades. For
example, advances in medicine and technology have increased life expectancy of many paediatric
conditions including asthma, leukaemia, sickle cell disease and cystic fibrosis (CF) (Perrin, Bloom &
Gortmaker, 2007). Such advances have caused a shift from short-term management of acute and
often fatal conditions to long-term management of survivable chronic conditions. Despite
improvement in life expectancy, paediatric chronic conditions can impose significant burden on
children, their parents, their families, and the public health system.
Integration of technology and science creating medical advancements has changed the
nature of many, once fatal, diseases to chronic conditions, insofar as a child who has a chronic
condition or disease in the modern era is expected to live a relatively ordinary life compared to 20 to
50 years ago. These changes in child health outcomes have significant and far-reaching impacts on
parental and family psychological adaptation to paediatric chronic conditions. Medical and
technological advancements have meant that more than ever, parents are finding themselves
providing all or most of the care for their chronically ill children. Parents are now being medicalised
(Valentine, 2010) in their efforts to keep their children healthy, by following treatment regimens and
obtaining knowledge of their child’s condition. Thus, in treatment of paediatric chronic conditions,
parents and family become focal points, as children are living longer and requiring extensive
treatment regimens to control symptoms. For these parents, their practices and their role can be
significantly different from parents who have healthy children.
Improvements in diagnosis and medical treatment have meant that children are being
diagnosed sooner, and as these improvements continue, their care is moving from the hospital
environment into the home and community. Though paediatric healthcare is improving, there is an
increased responsibility and burden on parents as primary carers of chronically ill children, which can
affect them in multiple ways. The first section of this chapter outlines what is currently known about
parenting children with chronic conditions, paying particular attention to theoretical understandings,
research trends and empirical outcomes.
Becoming a parent is often said to be one of the most life-changing experiences that
encompasses both stress and joy. The expectation is one of joy, stature and fulfilment whilst the
reality includes challenge, hardships and questioning one’s self as parent. Becoming a parent is
considered the most significant life-cycle transition in existence, and it is the most romanticised of all
the life transition stages (McGoldrick, Carter & Petkov 2011). Parenthood is common, with 299, 697
births registered in Australia in 2014 (Australian Bureau of Statistics, 2014). After the birth of an
infant it is the task and goal of parents to raise, protect and support the child through their early
years of life.
4 Parenting Children with Cystic Fibrosis
The transition to parenthood can be monumentally challenging when a baby is diagnosed
with a chronic life threatening condition. All parents hold expectations, hopes and dreams for their
child, but when a child is diagnosed with a chronic condition these aspirations are forever altered.
Learning of a child’s chronic condition is generally a devastating experience that usually sets in
motion a myriad of negative emotional responses. Whilst symptoms between conditions vary,
common factors have been identified in how parents react to their child’s diagnosis. Guilt, anger,
grief, shock and disbelief are common initial parental emotions to a child’s diagnosis (Fallowfield &
Jenkins, 2004; Goldbeck, 2006; Kars, Duijnstee, Pool, van Delden & Grypdonck, 2008; Moola, 2012;
Nguyen, Pertini & Kettler, 2013; Samson, et al., 2009; Willingham-Piersol, Johnson, Wetsel, Holtzer &
Walker, 2008). Most research into diagnosis of paediatric chronic conditions has focussed on the
mother because she has been identified as the one who generally carries the burden of care.
Research in this area has shown that exhaustion and frustration, loss of freedom and support
networks, and loss of ideal mother-child relationship are the main outcomes for mothers of a
chronically ill child (Almasri, et al., 2011; Bruce, Lilja & Sundin, 2016; Coffee, 2006; Esdaile &
Greenwood, 2003; Johnson, 2000; Lloyd & Rosman, 2005; Pelchat, Lefebvre & Perreault, 2003;
Shortman, et al., 2013; Wiedebusch, Pollmann, Siegmund & Muthny, 2008).
Early conceptualisations of parenting children with chronic conditions presumed that
children and their families would experience adverse psychological effects, referred to as
psychopathology, which in turn, was predictably found. For example, attachment and parenting style
issues (Eiser, 1993; Green and Solit, 1964; Marvin & Pianta, 1996, Venters, 1981), and depression
and chronic sorrow (Bowes, Lowes, Warner & Gregory, 2009; Cashin, Small & Solberg, 2008;
Johnson, 2000; Masterton, 2010; Swallow & Jacoby, 2001; Whittingham, Wee, Sanders & Boyd,
2013) have been commonly reported parental outcomes. Treatment and management of chronic
conditions, and its associated burdens has been a primary focus for clinicians and researchers alike,
with reports of anxiety, parenting stress and perceived parental incompetency in completing
required treatment regimens (Anthony, Bromberg, Gil & Schanberg, 2011; Barakat, Patterson, Daniel
& Dampier, 2008; Hall & Graff, 2011; Hassall, Rose & McDonald, 2005; Litzelman, Catrine, Gangnon
& Witt, 2011; Powers, et al., 2002; Shatla, El said Sayyah, Azzam & Elsayed, 2011; Waisbren, Wee,
Sanders & Boyd, 2004). These outcomes reflect the deficit-based models popular in the previous
decades of understanding parents, children and their families experiencing chronic disease.
Developmental pathway models have further conceptualised trajectories that result in
adaptive and maladaptive development. For example, McCubbin and McCubbin (1993) developed
The Resiliency Model of Family Stress, Adjustment and Adaptation that posited how families might
respond to a chronic condition within the family unit. Such developmental pathway models have
identified risk and resistance factors in managing paediatric chronic conditions, and have evolved
5 Parenting Children with Cystic Fibrosis
over time to direct what is used as a basis for intervention and promotion. Mechanisms that set
maladaptive trajectories have been categorised as risk factors, which are those elements within the
family’s environment, both inherent and indirect, that decrease likelihood of positive adaptation,
whereas resistance factors increase its likelihood (Gerhardt, Walders, Rosenthal & Drotar, 2004).
Researchers and clinicians alike have spent decades identifying these factors, their relationships with
other mechanisms and intervening where appropriate to obtain optimal adaptation to paediatric
chronic conditions.
Recent years have seen a tremendous growth and reconceptualisation in approaches to
understanding the impact of chronic disease on children and their families. The traditional deficit-
centred model has slowly been replaced with those models that take account of coping resources
and individual competence. The emphasis has shifted from identifying psychopathology within
children and their families towards an understanding of how ordinary children and their families deal
with specific crises. Increasingly, models are being drawn from mainstream psychology, with the
result that children and their families dealing with chronic conditions are not seen as
psychopathologic, but as ordinary people in exceptional circumstances (Eiser, 1990). For example,
research has shifted from identifying and intervening in maladjustment and psychopathology toward
identifying individual and family coping strategies (Varni & Wallander, 1988).
Research has shifted focus from psychopathologies of caring for a chronically ill child such as
depression and chronic anxiety to beneficial outcomes such as normalisation and resilience. For
example, a salutogenic understanding of existence focuses on factors that support health and well-
being, rather than on factors that contribute to adverse outcomes (Antonovsky, 1987). The aim of
such research is to anticipate and prevent dysfunction within families before its manifestation, by
strengthening parents and their families through providing education and support. Adaptive
behaviours are daily living skills that assist individuals and families to become productive within
society (Sparrow, 2011). Some resistance factors found to support adaptive functioning include
family support and resources, and normalisation of the illness and its management into the family
system. Support or resources that are currently used by the family, and additional supports
accessible to the family when a crisis occurs, have been reported as protective factors (McCubbin &
Patterson, 1983). Similarly, normalisation of a child’s condition and its treatment regimen into family
life can function as a resistance factor against practical, psychological and emotional burdens
associated with the condition (Hopkins & Gallo, 2012; Knafl, Darney, Gallo & Angst, 2010; Ray, 2002).
Accordingly, by its very nature, normalisation of paediatric chronic conditions into the family context
may buffer and protect its members against their existence as well as against future burden, both
normative and condition-specific.
6 Parenting Children with Cystic Fibrosis
Coinciding with a shift from deficit-based models to salutogenic approaches (Antonovsky,
1987) of research and clinical care, is increasing acknowledgement of the relationships between an
individual’s psychosocial environment and their health and well-being. Importance of family context
for child health is emphasised by a biopsychosocial understanding of health, which recognises that
the psychosocial environment of the child is a critical factor in the health of a child with a chronic
condition (Engel, 1977; 1980). The family is the most important influence within the psychosocial
environment of the child (Bronfenbrenner, 1986). Increasing both life expectancy and physical
health outcomes are the primary foci of biomedical innovation, research and clinical care. However
the biopsychosocial model of care (Engel, 1977; 1980) highlights importance of a holistic approach to
an individual’s care, inclusive of psychosocial and physical health and well-being of the child and
their family.
Engel (1977; 1980) claimed that not only biological factors are important in individual
functioning in the context of disease or illness. He claimed that psychological and social factors are
also important considerations, both in terms of impact and outcomes of disease or illness. Engel’s
biopsychosocial model of disease and illness echoes a social-ecological perceptive (Bronfenbrenner,
1979; 1986) to approaching human functioning in the context of disease or illness through its
emphasis on interactions between biological, psychological and social factors. This contrasts the
more traditional biomedical model of medicine that suggests that every disease process can be
explained in terms of an underlying biological deviation from normal functioning (Stedman, 2011).
Since Engel’s (1977; 1980) assertion that not only biological factors should be considered in
care of sick individuals, but that psychosocial factors may impact on the biology of an illness, social-
cognitive models of health behaviour have been formulated and tested. With the model’s social-
cognitive underpinnings, acceptance of such models varies across cultures (Santrock,
2007).Recognition of the biopsychosocial model in clinical care is evident in the United States, the
United Kingdom, Australia and some European countries where integration of professional health
services aims to provide care and address a patient’s needs on all three levels (Gatchel & Oordt,
2003).
Over the past 20 years, qualitative studies have conceptualised family response to paediatric
chronic conditions, and quantitative studies have explained the nature of family response to illness
or identified individual-level variables that explained a family’s response to illness. Descriptions of
families’ responses to illness include family life in the context of illness, impact of illness on the
family system, family transitions related to illness and development of family concepts in context of
the illness (Rolland, 1984, 1987). Explanations of responses to illness within families can be predicted
by such factors as cohesion and conflict. For example, the responses of individual family members to
7 Parenting Children with Cystic Fibrosis
illness and interventions influence family functioning as a whole, which in turn can inform or modify
functioning of each individual (Walsh, 2003).
The shift to a family focus of child health has encompassed embracing the fathers’ roles in
their child’s care. This shift coincided with modern arrangements of family systems and child care
responsibilities, resulting in fathers becoming more involved in their child’s care. Whereas once,
identification of mother as primary caregiver meant that aims and principles focussed on her
psychological and social well-being, and her relationship with the child and other family members.
The dominant or defining structure of paternal parenting has shifted in succession from an emphasis
on moral guidance, to a focus on breadwinning, then to sex-role modelling, marital support, and
finally, nurturance (Lamb, 2000). Traditional parental roles were gaining popularity, prompting a
restricted focus on paternal nurturance and involvement in day-to-day child care, with little, if any,
attention paid to the other functions or aspects of fatherhood. Consequently, academia has only
recently begun to seek a broader and more inclusive understanding of fatherhood. These efforts
have permitted more insightful research on the motivations and behaviour of contemporary fathers,
as well as on the effects of variations in paternal behaviour on child health and development (Lamb,
2000).
Though fathers’ roles and experiences are under-represented in the literature, the need to
consider fathers, along with their challenges and benefits in caring for a chronically ill child are
increasingly recognised (Phares, Lopez, Fields, Kamboukos & Duhig, 2005; Tiedje & Darling-Fisher,
2003). Like mothers, fathers can experience stress (Calzada, Eyberg, Rich & Querido, 2004; Weiner,
Vasquez & Battels, 2001) and post-traumatic stress syndrome (Ribi, Vollrath, Sennhauser, Gnehm &
Landolt, 2007). Fathers can experience difficulty discussing and expressing their family situation
(Goble, 2004), which can be compounded by their own expectations that they should be strong and
silent. The interaction between stress and gender can challenge the fathers’ ability to cope
(Arockiasamy, Holsti & Albersheim, 2008), and by remaining strong and silent, fathers are less likely
to receive support (Clark & Miles, 1999). This perception of needing to portray strength within the
family unit may also impede the fathers’ involvement through desire to maintain control whilst at
the same time feeling a loss of control (Clark & Miles, 1999; Hovey, 2005). In fact fathers can, for the
most part, feel like a forgotten parent because they are often not involved in doctor’s appointments
or the child’s treatment regimen (Sterken, 1996). Lastly, fathers’ can sense incompetency in their
child’s care because of their perceptions of the mother as expert in care for the ill child, which can
result in low self-esteem (Katz & Krulilk, 1999).
Similar to the shift from an exclusive focus on mothers as important people in the care of
chronically ill children, there was a shift from an exclusive biomedical to a biopsychosocial view of
care (Engel, 1977; 1980). A concurrent shift from a psychopathologic, deficit-based to a salutogenic
8 Parenting Children with Cystic Fibrosis
(Antonovsky, 1987), strengths-based perspective of care resulted in recognition of family resilience
as an important buffer against future burden of a child’s condition, as well against other life
stressors (Benzies & Mychasiuk, 2009; Black & Lobo, 2008; Patterson, 2002; Walsh, 2003). The shift
to a salutogenic approach, along with increases in life expectancy has shed new light on parenting a
child with a chronic condition. These insights give hope for parents, children and their families that
have not previously existed.
When a child is diagnosed with a chronic condition and life expectations are irrevocably
altered, hope can take its place so that an alternative reality can become positively reframed. Hope
in the context of parenting a child with a chronic condition has been referred to as a transitional
refocusing from a difficult present to a positive future with dynamic possibilities within continual
uncertainty (Duggleby, et al., 2010). Hope has been reported by parents of children who have
various chronic conditions. Parents have hope for their child’s; quality and quantity of life (Lloyd &
Hastings, 2009), employment, family and other normative life transitions (Samson, et al., 2009) and
they even hope for a cure to the condition for which their child suffers (Wong & Heriot, 2008). Hope
can contribute to resilience for future burden of physical and mental chronic conditions (Faso, Neal-
Beevers, & Carlson, 2013; Horton & Wallander, 2001; Kashdan, et al., 2002; Lloyd & Hastings, 2009).
Therefore, the salutogenic (Antonovsky, 1987), along with the biopsychosocial (Engel, 1977; 1980)
perspectives support the notion that hope within the family context is a highly regarded parental
trait. The biopsychosocial (Engel, 1977; 1980), along with the social-ecological (Bronfenbrenner,
1979; 1986), perspective on caring for children who have chronic conditions emphasises the
importance of parent and family practices and outcomes for child health outcomes.
A child’s experience with their immediate environment provides the building blocks for their
growth and development. Neurobiological effects of external psychosocial factors on child
development, that is, psychosocial factors within the environment of the child have been shown to
affect plasticity of the brain during windows of opportunity. For example, synapse formation and
pruning during the early years of life are influenced by the child’s environment. This has been
demonstrated whereby environmental stimulation can positively and negatively affect brain
development in the early years of life through effects on neurogenesis, synaptogenesis, long-term
potentiation and pruning. These findings have been demonstrated at the cellular level, in vitro and in
humans (Jontes, Buchanan & Smith, 2000; Mirescu, Peters & Gould, 2004; Stanwood & Levitt, 2004;
Van Praag, Christie, Sejnowski & Gage, 1999). For this reason, early childhood experiences affect
gene expression and therefore affect developmental and long-term health outcomes. Accordingly,
the first 1000 days of a child’s life, starting from conception to their age of 2 years, are important for
all domains of development (Hoddinott, et al., 2013).
9 Parenting Children with Cystic Fibrosis
Social-ecology theory posits a relationship between the developing child and the settings
and contexts in which the child is actively involved. Bronfenbrenner (1979) proposed a model of bi-
directional influences on the child within their environment as occurring within nested settings
beginning with the child, microsystem, and extending out to the immediate social settings of home,
school and neighbourhood, mesosystem, and settings that do not involve the child as an active
participant such as the parents’ workplace, the exosystem, and the wider society and culture,
macrosystem. A substantial body of literature supports utilisation of the model for families of young
children, suggesting that indirect influences on child development can be traced back to parental
characteristics (e.g., Armstrong, Birnie-Lefcovitchb & Ungar, 2005; Brophy-Herb, Lee, Nievar &
Stollak, 2007; DePanfilis & Dubowitz, 2005; Dishion & Stormshak, 2007). Consequently, based on
understandings of social-ecological theories, a major line of inquiry has been to determine if and
how parenting behaviours and parental characteristics influence child development.
Using this framework, parenting has been well-established as an important influence on
child development, and much research has linked parenting with both beneficial and adverse child
outcomes (e.g., Baumrind, Larzelere, & Owens, 2010). Research has shown parenting behaviours and
parental characteristics can influence academic achievement (Cheung & Pomerantz, 2011; Englund,
Luckner, Whaley, & Egeland, 2004, Pomerantz, Moorman, & Litwack, 2007), cognitive development
(Ryan, Martin, & Brooks-Gunn, 2006), socio-emotional development (Bernier, Carlson, & Whippe,
2010; Dallaire, et al., 2006; El Nokali, Bachman, & Votruba-Drzal, 2010), and school dropout rates
(Marcus & Sanders-Reio, 2001). Research has shown parenting can influence cognitive development
(Ryan, et al., 2006), and socio-emotional development (Bernier, et al., 2010; Dallaire, et al., 2006; El
Nokali, et al., 2010) in infants and young children. Moreover, family psychosocial functioning,
including parenting, is well known to impact normal child development (Beauchamp & Anderson,
2010).
Based on social-ecology principles, parents play an integral role in their child’s ability to
adapt to living with a chronic condition, not only in terms of their child’s developmental functioning
but also in their ability to participate in daily activities. Principally, parent psychological distress has
been identified as a risk factor for adverse outcomes in children with a variety of chronic conditions
including CF (Cappelli, McGrath, MacDonald, Boland & Katsanis, 1988), cancer (Robinson, Gerhardt,
Vannatta & Noll, 2007), type 1 diabetes (Whittemore, Jaser, Chao, Jang & Grey, 2012), spina bifida
(Freidman, Holmbeck, Jandasek, Kukerman & Abad, 2004) and chronic pain (Palermo, Putnam,
Armstrong & Daily, 2007). Therefore, parents have significant potential to beneficially or adversely
affect their child’s adaptation to their chronic condition (Law, Fisher, Fales, Noel & Eccleston, 2014).
A child’s adaptation to their condition then has both direct and indirect implications for their own
10 Parenting Children with Cystic Fibrosis
development. For example, direct implications include psychological and emotional well-being
whereas indirect implications include treatment adherence.
Parenting is now well-established as an important influence on child development with
extensive research linking parenting with beneficial and adverse child mental health outcomes. To
that end, understanding parenting; its process, functions and outcomes, and parental characteristics
and their outcomes are not only important for parents’ general health and well-being per se, but it is
now well-known that parents matter when it comes to their children’s development and long-term
health outcomes. Therefore, parenting research and intervention in the early years of a child’s life
will most likely beneficially impact upon the entire family system.
1.1.2 Paediatric Cystic Fibrosis in Australia
Cystic fibrosis (CF) is a complex, chronic disease for which there is currently no cure.
However, life expectancy has exponentially increased over the past few decades, changing CF from a
fatal childhood digestive and lung disease to a complex multi-systemic disease extending into
adulthood (Davies, Alton & Bush, 2007). Advances in science, medicine and technology have resulted
in life expectancy of CF patients increasing from 5 to 6 years in 1960 to a median survival age of
approximately 40 years now (UK Cystic Fibrosis Registry, 2012). Improved outcomes for children
born with CF are attributed to early detection and diagnosis through newborn screening (NBS)
programs and improved methods of treatment and management (e.g., Farrell, et al., 2001; Farrell, et
al., 2005).
Cystic fibrosis is an autosomal recessive disease most commonly affecting Caucasian people
(Davies, et al., 2007; Montgomery & Howenstine, 2009) that primarily affects the gastro-intestinal
system, respiratory system, and sweat glands (Davis, 2006; Rowntree & Harris, 2003). Approximately
70% of children with CF are diagnosed before the age of 1 year (Walters & Mehta, 2007), with
clinical symptoms often present soon after birth (FitzSimmons, 1993). Data registry services in
Europe, the United Kingdom, the United States and Australia collectively report over 10,000 children
under the age of 7 years are living with CF (Cystic Fibrosis Australia, 2013; Cystic Fibrosis Foundation,
2014; European Cystic Fibrosis Society, 2014; UK Cystic Fibrosis Registry, 2013)
The generally overwhelming and demanding nature of CF on the mental health and well-
being of parents likely explains why modern research still primarily focusses on psychopathology of
children and their parents. A large epidemiological study currently being conducted across the
United States, the United Kingdom, Europe and Australia is investigating stress, depression and
anxiety for children with CF and their caregivers. Preliminary findings report that approximately a
quarter of the caregivers sampled reported clinical levels of stress, depression and/or anxiety
(Besier, et al., 2011; Driscoll, Montag-Leifling, Acton & Modi, 2009), an already well-known finding
(Goldbeck, Quittner & Besier, 2008; Quittner, et al., 2008; Yilmaz, et al., 2008).
11 Parenting Children with Cystic Fibrosis
Two particular aspects of CF care are important in understanding how parents manage their
child’s CF treatment, as well as how they experience their child’s condition. Firstly, treatment
management primarily occurs within the home. Like some other chronic conditions that were once
considered fatal (e.g., chronic kidney disease), parents whose children have CF are required to take
primary responsibility for their child’s treatment management, which is generally considered to be
demanding and time-consuming. Therefore, treatment burden is reportedly common amongst
parents, which can include parenting stress and parental role strain associated with treatment
administration (Hodgkinson & Lester, 2002; Quittner, Opipari, Regoli, Jacobsen & Eigen, 1992;
Quittner, et al., 1998).Sometimes treatment burden is a result of low levels of perceived
competence with administration of therapies (Sawicki & Tiddens, 2012), and anxiety associated with
treatment burden can occur because of increased awareness of disease trajectory (Sawicki, Hellar,
Demars & Robinson, 2015). Secondly, parents find it difficult to obtain peer support from other
parents experiencing the same situation because children with CF should not communicate face-to-
face with each other due to issues of cross-infection. Peer support from parents whose children have
the same condition is an important factor (Duffy, 2011; Eatough, et al., 2013; Kerr, Harrison, Medves
& Tranmer, 2004; Shields, Young & McCann, 2008) and could be an essential component of parental
adaptation to a paediatric chronic condition (Barcroft, 2015).
Traditionally in CF, the early years of life have been considered the silent years. This is
because disease progression is usually asymptomatic and detection using traditional methods, such
as lung function, is difficult as young children struggle to cooperate with the demands of the
investigations. Parental reactions to, and experiences of, diagnosis have been thoroughly
investigated (e.g., Baroni, Anderson & Mischler, 1997; De Monestrol, Brucefor, Sjoberg & Hjelte,
2011; Havermans, Tack, Vertommen, Proesmans & de Boeck, 2015; Quittner, DiGirolamo, Michel &
Eigen, 1992). As too have parental responses and experiences of disease progression and treatment
management later in a child’s life (e.g., Dashiff, Suzuki-Crumly, Kracke, Britton & Moreland, 2013;
Fidika, Herle, Herschbach and Goldbeck, 2015; Sawicki, et al., 2015), usually from approximately 10
years of age when the condition generally starts to become symptomatic. However the mental
health and experiences of parents with young children who have CF diagnosed early by NBS has not
generally been considered.
Following the challenge of diagnosis and responsibility of home treatment is the awareness
that CF disease progression is occurring, often without symptoms and in children who look well. Due
to medical and technological advancements, parents are receiving more detailed information about
their child’s condition and at a much earlier time than ever before. Due to the recentness of such
advancements in treatment and detailed information about disease progression being generated,
how parents respond to, and manage, such information remains largely unknown.
12 Parenting Children with Cystic Fibrosis
1.1.3 Current Australian treatment management practices for infants and young children
in the 21st century
Much of the medical care for individuals living with CF in Australia is provided by specialist
CF centres peer-reviewed by the national CF body – Cystic Fibrosis Australia (CFA). Australian
national standards of CF care were developed in 2008 and promote standardised care across centres
(Bell & Robinson, 2008). Cystic fibrosis centres provide a multidisciplinary allied health and medical
team with facilities for relevant diagnostic testing and other subspecialist medical care (Bell 2008
Schechter & Gutierrez, 2010). A holistic approach to CF care is fundamental, and care is delivered by
a multidisciplinary team of health professionals with expertise in CF (Madge & Khair, 2000). The
main aims of CF care include preventing chronic infection, minimising progression of lung damage,
sustaining normal nutrition and growth, maintaining independence for patients and their families,
and maximising quality of life (Agent & Madge, 2007). Major CF centres provide care for local
patients with CF, as well as those patients who live regionally via outreach programs (Littlewood,
2000).
The people who form a typical CF multidisciplinary team include lead clinical physician, and a
specialist clinical nurse, physiotherapist, dietitian, psychologist, pharmacist, and social worker.
Access to further multi-disciplinary team members including specialised laboratory services,
gastroenterologist, diabetiologist, and an ear, nose and throat specialist (Agent & Madge, 2007) is
essential. The CF multi-disciplinary team has expert knowledge in CF and provide clinical,
educational and support services for patients and their families (Agent & Madge, 2007). The
specialist nurse has a unique role by being involved with the patient and their family over the
lifespan: from helping them come to terms with the CF diagnosis to supporting them with end-of-life
management (Agent & Madge, 2007). Currently, clinical guidelines and standards of care are
generally limited to medical aspects of CF, with much less consideration of accompanying
psychosocial aspects (Colombo & LIttlewood, 2011; Tiddens, 2009). However, Borowitz, et al., (2009)
recently developed Cystic Fibrosis Foundation evidence-based guidelines for management of infants
with CF. Furthermore, a European and American collaborative consortium recently advocated for
screening anxiety and depression in patients and their caregivers, making recommendations for
international implementation (Abbott, et al., 2015; Quittner, et al., 2015).
As part of standard paediatric CF care in Australia, an annual review is completed for each
child, which consists of a comprehensive assessment by the multidisciplinary team of all aspects of
their condition and treatment (Agent & Madge, 2007). The aim of the annual review is to gain a
thorough understanding of all aspects of a child’s condition, identification of suboptimal treatment
that can be imporved, formalisation of an individual management plan; and provision of information
13 Parenting Children with Cystic Fibrosis
for centre and national databases (Agent & Madge, 2007). Important aims of the review are to
inform parents of the progress of their child and provide a care plan for the next 12 months.
The annual review consists of a medical history review, assessment of CF by a specialist
clinical nurse and specialist physiotherapist, nutritional assessment by a specialist dietitian, review
by a psychologist and social worker, and full clinical examination (Agent & Madge, 2007). The annual
review for children up to 7 years of age attending paediatric CF centres at Princess Margaret Hospital
(PMH) and The Royal Children’s Hospital (RCH), is unique in Australia and much of the world in that it
includes pulmonary surveillance using the most detailed measures of lung disease currently
available. These investigations provide information regarding the structural, microbiological and
inflammatory health of the lungs. Research that is focused upon improving the physical and mental
health outcomes of children with CF is embedded in the clinical program and is further supported by
collaborations with teams in Chapel Hill, North Carolina, Indianapolis, Indiana, St Louis, Washington
and Rotterdam, Netherlands.
As standard treatment for CF, parents must perform several hours of therapy a day in the
home such as preparing a special diet, administering medications, completing airway clearance with
physiotherapy, and performing nebulisation therapy so that aerosol particles can be breathed
directly into the lungs (McCubbin, Bowers & Holaday, 1984). When a child with CF is in good health,
treatment may take a few hours a day, but when they become ill, treatment increases substantially
(Agent & Madge, 2007). Promotion of airway clearance of mucus, suppression of bacterial growth
through prophylactic antibiotics and optimisation of nutritional status through pancreatic enzymes
are the main pillars of CF management (Davies, et al., 2007; Montgomery & Howenstine, 2009). Due
to the inability of the lungs to clear thick mucus secretions that contain a number of bacteria and
other airborne particulates, airway clearance techniques are a standard part of CF care (Quittner,
Barker, Marciel & Grimley, 2009). Chest physiotherapy loosens mucus in the lungs and encourages
its removal though productive coughing. This process can take up to 1 hour and is typically
performed twice a day (Quittner, et al., 2009).
Despite such an aggressive and proactive treatment regimen, fighting lung infection is a
lifelong battle with the lungs eventually becoming chronically infected with bacteria, such as
Pseudomonas aeruginosa. Treatment of lung infection requires oral and nebulised antibiotics in
many patients: an additional 20 minutes of treatment daily (Collins, 2009). If deterioration of the
patient’s respiratory condition (that is, a pulmonary exacerbation) continues despite these
treatments, intravenous antibiotics can be prescribed for a period of 10 to 14 days (Quittner, et al.,
2009) for which a hospital admission is generally required.
The digestive system of people with CF is also affected, with most infants (85%) born with
pancreatic insufficiency (Zielenski, 2000). This refers to the impediment of the pancreas to secrete
14 Parenting Children with Cystic Fibrosis
enzymes necessary to digest food resulting in its inability to effectively absorb food (Montgomery &
Howenstine, 2009). This problem is addressed by consuming orally administered enzymes with every
snack and meal. Malabsorption and increased resting energy expenditures are issues in CF, requiring
patients to consume 100 to 200% of the recommended daily allowance of calories (Stallings, et al.,
2008). Therefore, children can experience issues gaining weight if they do not maintain a high-fat
diet (Ramsey, Farrell & Pencharz, 1992). Somatic growth and development of children with CF is a
concern related to both the gastrointestinal and respiratory problems associated with CF.
1.1.4 Cystic fibrosis treatment management in infants and children from diagnosis to 7 years in
Western Australia and Victoria: Australian innovation for paediatric CF management
The introduction of NBS to Australia over 20 years ago has resulted in early diagnosis and a
paradigm shift from reactive treatment to proactive early-life disease surveillance and therapeutic
intervention. The Australian Respiratory Early Surveillance Team for Cystic Fibrosis (AREST CF -
www.arestcf.org) pioneered early detection and prevention of CF disease progression, and is a multi-
disciplinary clinical and research collaboration between paediatric CF centres in Perth, Western
Australia and Melbourne, Victoria who are dedicated to improvement of respiratory health and
outcomes in children with CF. The program was initiated at Princess Margaret Hospital (PMH) in
Perth in 1996 to investigate the role of inflammation in the lungs of children with CF. Since then, the
program has grown into a comprehensive assessment of early life disease in CF. Bronchoalveolar
lavage (BAL) was introduced in 1999, it measures inflammation and infection in the lung via a
bronchoscopy. Infant and preschool lung function testing was introduced in 2003 and chest
computed tomography (CT) scanning to define lung structure was introduced in 2005. All BALs and
CTs for infants and young children are performed under general anaesthetic (GA). The Royal
Children’s Hospital (RCH) in Victoria joined the program in collaboration in 2005. The main objective
of the AREST CF team is to improve the detection, prevention and treatment of early respiratory
disease in young children with CF in order to improve clinical outcomes and quality of life for
patients and their families.
Central to AREST CF is the early surveillance program: a unique clinical surveillance program
that has the potential to modify long term outcomes for children with CF through early detection
and interventions to delay the onset of lung disease, long before signs and symptoms are apparent.
The program is aimed specifically at infants and preschool children from diagnosis (approximately 3
months) to 7 years who undergo annual, detailed surveillance procedures, which are relatively
invasive compared to standard practices in Australia. These investigations generate detailed
information about the progress of early lung inflammation, infection and structural disease that
would otherwise remain largely unknown to the treating physicians and the families. Whilst early
surveillance is currently unique to Perth and Melbourne, it is rapidly emerging as the likely
15 Parenting Children with Cystic Fibrosis
framework for future therapeutic intervention trials in young children nationally and internationally.
Currently, the surveillance framework has been adopted by an international clinical trial (Australia,
New Zealand, Netherlands), COMBAT-CF, which uses bronchoscopy and chest CT scans as outcome
markers of CF disease progression. Since its inception, approximately 95% of Western Australia’s and
Victoria’s populations of children with CF are enrolled in the program from diagnosis. Following is a
detailed description of each of the procedures involved in the AREST CF early surveillance program.
Bronchoalveolar Lavage
Bronchoalveolar lavage is performed under the guidance of a bronchoscope. A
bronchoscopy and BAL are performed in children who cannot yet cough up sputum. Older
children can do this, which can then be tested for infections. Young children find this difficult to
achieve, and as the children in the program are under the age of 7 years, this technique is
appropriate. BALs consist of a GA administered and positioning of a disposable laryngeal mask
airway (LMA) to facilitate passage for the bronchoscope (Stick, et al., 2009). The bronchoscope is
passed via the LMA through the vocal cords into the trachea and down into the first and second
main bronchi to reach the lobe where BAL is performed (Brennan, Gangell, Wainwright, & Sly,
2008). A bronchoscope is a fibre-optic tube with a camera at the end, which allows clinicians to
investigate airways in the lungs. A BAL involves instilling saline into a small part of the lung and then
removing it through suction. The returned fluid collects mucous located in the area of the lung
where the fluid was inserted. The fluid is then assessed for bacteria or inflammation in the lungs.
BAL is performed using normal saline warmed to body temperature (Brennan, et al., 2008). Both
bronchoscopy and BAL are considered the gold standard for determining airway inflammation and
lower respiratory tract infection (Ranganathan, 2006), and BAL is currently the only method for
obtaining reliable data on lower respiratory tract infection in young children with CF (Brennan, et
al., 2008).
Lung Function
Lung function assessment in young children undergoing early surveillance consists of a
number of different techniques depending on the age of the child. Lung function can be
assessed similarly in infants and preschool children but with slightly different techniques. There
are three different lung function tests for infants up to 2 years of age. The tests take
approximately 3 hours in total and are performed the week prior to the CT scan and BAL. As infants
are unable to provide the cooperation required for typical lung function tests, specialised methods
have been developed to assess lung function in this age group. That is, tests have been modified so
that large, forced breaths are not required by infants, or the machines perform this technique for
the infant. Infants are given a sedative, chloral hydrate, prior to commencement of the tests.
16 Parenting Children with Cystic Fibrosis
Multiple breath washout testing is designed to determine how evenly gas mixes in the lungs,
and involves the infant breathing in gas that contains a known concentration of an inert tracer. The
concentration of tracer gas in exhaled air is monitored during tidal breathing. The test provides an
assessment of ventilation inhomogeneity and lung clearance index arising from mucus plugging of
the small peripheral airways and airway wall thickening (Rezaee & Ren, 2006). Low-frequency forced
oscillation is another technique that measures total respiratory impedance by measuring changes in
pressure and flow in response to a low amplitude oscillatory pressure signal applied at the airway
opening (Oostveen et al., 2003; Ranganathan, Linnane, Nolan, Gangell, & Hall, 2008).
The last test is the raised volume rapid thoraco-abdominal compression technique, which
assists in determining obstruction of air flow in the lungs. Prior to starting the test procedure, the
infant is wrapped in an inflatable jacket, and the infant’s lungs are gently inflated with a pump.
Once the lungs are filled to a set inflation pressure, the jacket is rapidly inflated, squeezing the
thorax, which simulates a forced expiration (LeSouef, Turner, & Motoyama, 1996). Obstruction of
airflow is measured by the amount of air that is expelled in the forced expiration.
Children over 2 years of age perform slightly different tests based on their ability to expel air
from the lungs on demand, and are not sedated for lung function testing. Young children perform
the multiple breath washout procedure and forced oscillation technique. However, this is not
conducted under low frequency with young children and is performed using a slightly different
technique. The child is required to breathe normally through a mouthpiece whilst wearing a nose
clip. A low amplitude pressure oscillation is applied at the airway opening. This test is a measure of
airway resistance and lung stiffness.
Young children 5 years of age and older also perform spirometry (forced expiration), which
results in a measure of FEV¹ (a measure of airway obstruction). This test requires a maximal
expiratory maneuver involving an inspiration to total lung capacity before a forceful exhalation to
residual volume (Mayer & Allen, 2006). The test provides information about airway obstruction and
is used as a standard measure of lung function in older children and adults (Kerem, Conway, Elborn
& Heijerman, 2005.
Computed Chest Tomography
Lung function assessments are provided through CT. Chest scans provides imaging of the
lung that shows the presence, severity and extent of lung disease. Previously, physicians have
been limited to using chest x-rays, but advances in techniques now enable more
comprehensive assessment of the lungs. Chest scanning is an imaging technique that is more
detailed than chest x-rays in providing information on structural C F abnormalities, especially
in young children. Chest scans are performed annually under GA. The CT scan is performed under
sedation as precise volume control is required and infants and young children who aren’t able to
17 Parenting Children with Cystic Fibrosis
cooperate (Kirks, 1998). The CT procedure has been modified to minimise exposure to ionising
radiation, a common concern with imaging techniques. Currently there is international effort to
develop a standardised low-dose CT protocol suitable for children with CF (Linnane, Robinson,
Ranganathan, Stick, & Murray, 2008). Chest scans are considered the gold standard technique in
diagnosing bronchiectasis (Chang & Redding, 2006), and has the ability to clearly determine
structural components of CF lung disease (Oikonomou & Hansell, 2006; Sonnappa & Owens, 2007).
All BALs and chest CTs for those infants and young children under 7 years of age are
performed under GA. Anaesthesia is induced with either sevoflurane or propofol, according to the
clinical judgment of the anaesthetist (Stick, et al., 2009). Parents can stay with their infant or child
for the duration of the lung function tests and whilst their infant or child is being anaesthetised.
Parents must, however, leave theatre for the bronchoscopy and BAL procedures, and the CT scan
due to ionising radiation. Parental participation during testing is not encouraged due to the
complexity of the procedures. All procedures are generally scheduled as a day surgical case with the
child discharged the same day (Brennan, et al., 2008).
A week in the life of parents involved in AREST CF during annual surveillance
Parents of infants up to 2 years of age
In the week preceding coming into the clinic for their infant’s CT and BAL, parents bring their
infant in for a multiple breath washout procedure, low frequency forced oscillation procedure and
raised volume rapid thoraco-abdominal compression procedure. These procedures are performed
on the same occasion and under sedation. With much variation within the amount of time it takes
for the infant to fall asleep with sedation, for the tests to be completed and for the infant to fully
recover from sedation, the average time parents are required to relinquish their infant is
approximately three hours.
Parents of children aged 2-7 years
Children aged 2 years and older undergoing early surveillance perform forced oscillation
technique and spirometry, as well as CT and BAL conducted under GA. This requires a full day of
testing for children, and waiting for parents. The surveillance procedures for all children within the
program are performed in addition to 3-monthly clinic reviews, which include height and weight
measurements, social worker, physiotherapist, dietician and physician appointments.
In summary, this introduction has focused on the research context, specifically: CF in young
children and treatment required in maintaining their optimal health; what parents experience and
how they cope with their child’s chronic condition; and what the AREST CF early surveillance
program offers children with CF and their parents, along with the processes and procedures
involved. How these contextual factors have informed the framework, foundation and purpose of
this research has also been discussed. The following section provides a summary of the research
18 Parenting Children with Cystic Fibrosis
rationale and outlines specific aims and questions of my research. The last section concludes by
highlighting the contribution of my research to contemporary understandings of children’s and
families’ involvement in early surveillance for covert (i.e., undetected by traditional measures) CF
disease progression, in particular, by highlighting the significance of the research for policy, practice,
research and theory.
1.2 The current research
1.2.1 Research rationale
Despite the breadth of debate, as well as decades of clinical and theoretical awareness of
the psychological needs of parents who have a child with a chronic condition, no research was found
that specifically investigated the experience of parenting children with CF in the context of early
surveillance for CF lung disease. The necessity to address this paucity in research was emphasised by
theoretical understanding of the parenting experience that has been developed without
consideration of processes and outcomes of parenting in the context of early surveillance for
paediatric chronic disease. Given the current clinical and political agendas for early intervention
(Jenkins, 2005; Wise, da Silva, Webster & Sanson, 2006), there has been a need for research into
parental experiences of early intervention and aggressive treatments. Further emphasised by policy
and program development and implementation is the need to understand parents’ experiences and
how they cope with early intervention and aggressive treatment for their children.
Further is the increasing recognition that if research is to become meaningful in public
health systems and the provision of health care services, it must be translational (Oborn, Barrett &
Racko, 2010; Ogilvie, Craig, Griffin, Macintyre & Wareham, 2009). Although no single definition of
translational research has been agreed upon, it has been described as a necessity to translate
knowledge created in the research setting into real-world applications that aim to improve health
and well-being of individuals through provision of more effective health care systems and services
(Canadian Institutes of Health Research, 2004). A fundamental rationale underpinning the view on
translational research by the Canadian Institutes of Health Research (CIHR) is a sense of moral and
social responsibility to ensure that research progresses such that individuals who funded and enable
health research in the first place benefit from its conduct. Therefore, for research that is
fundamentally explorative to become translational, it must consider aspects of human experience
unreachable by positivist methods so that humanistic phenomena uncovered by the research can be
addressed.
Parents of children diagnosed with CF via NBS to 7 years of age and currently under
surveillance for lung disease, were targeted as research participants. The purpose of my research is
to explore how parents of children undergoing early surveillance for CF lung disease construct their
lived experiences of parenting, and how they attribute meaning to these experiences, drawing
19 Parenting Children with Cystic Fibrosis
attention to how they cope. A further aim of my research is to contribute to developing a modern
support system. .
Accordingly, to support parents in the AREST CF program, it is important that current
understandings of their experience be reviewed with reference to the lived experience of paediatric
chronic disease. By asking parents not only about how they construct their experiences of parenting
a child with a chronic condition but also examining how they attribute meaning to their experiences
is required so that more meaningful analysis of theory, policy and clinical practice effectiveness can
occur. My research aims to achieve this by exploring constructions of experiences and attributions of
meanings from the perspectives of those parents involved in the AREST CF program. Information
about the lived experience of parenting was explored with interview data from both sites where
AREST CF operates in Australia. The sample was stratified into two groups by child age. The
relevance of sampling across both sites and stratifying by child age was such that diverse
experiences could be interrogated, and changes that parents experienced over time could be
identified. The next section outlines what is currently known about parenting children with chronic
conditions who undergo early surveillance for disease progression.
1.2.2 Exploring the lived experience of parenting children with CF in the context of early
surveillance
With the successive, yet sporadic, implementation of NBS throughout the world there is now
considerable research that addresses psychosocial effects of NBS and diagnosis. However, research
characterising the psychological effects of early CF disease surveillance (particularly programs that
use relatively invasive modalities for disease detection) following NBS does not exist, nor does
literature exist on how children with CF, their parents and families experience, or cope with, early
disease surveillance like that operated in the Perth and Melbourne paediatric CF centres. Therefore,
little is known about what parents experience and how they cope with a previously untested
program of management in young children with CF and what factors are associated with adaptive
and maladaptive coping. Moreover, it is not yet clear if there are psychological sequelae of early
surveillance and early (often covert) disease detection.
Anecdotal experiences and assumptions of healthcare professionals (HCPs) working within
the program are that parents seem to adapt to the program over time, although clearly some
parents experience, and cope with, the program better than others. Professionals conducting early
disease surveillance programs following NBS diagnosis need to; understand whether or not families
perceive the program to be adversely affecting their lives; and identify psychological characteristics
within the family that may be associated with adjustment and maladjustment or other positive and
negative experiences. Consequently, care pathways for improved mental health and well-being for
families involved in early surveillance are incomplete. Therefore my research should provide
20 Parenting Children with Cystic Fibrosis
empirical support for anecdotal experiences and assumptions of HCPs working with families and may
provide some understanding of psychological and emotional processes and outcomes for parents
whose children undergo early surveillance for CF lung disease, which could guide future clinical
interventions and facilitate further psychological research.
1.2.2.1 Parallels drawn from medical treatment procedures.
As there is no research assessing parental psychological effects of the AREST CF program, it
is logical to consider research assessing similar parental experiences found within the program, as
this will provide the closest idea of what parents may experience based on similarities, for example,
in medical procedures. Therefore, anticipated experiences can be drawn from the literature in other
diseases that require invasive procedures as part of care. These experiences include anxiety
(Bauchner, Vinci, Bak, Pearson, & Corwin, 1996; Besier, et al., 2011; Chundamala, Wright, & Kemp,
2009; Menahem, Poulakis, & Prior, 2008), stress (Da Silva, Jacob, & Nascimento, 2010; Hopia,
Tomlinson, Paavilainen, & Åstedt-Kurki, 2005; Hutchinson, Willard, Hardy, & Bonner, 2009), need for
control over their child’s care during hospitalisation (Berrios-Rivera, Rivero-Vergne, & Romero, 2008;
Brosig, Mussatto, Kuhn, & Tweddell, 2007; Hallstrom & Runeson, 2001), and role strain (Quittner, et
al., 1998; Quittner, et al., 1992). Repeated exposure to these experiences in the absence of supports
may reflect adversely on the well-being of the family and crucially affect the child (Patterson, Budd,
Goetz, & Warwick, 1993; Szyndler, Towns, Van Asperen, & McKay, 2005; Turner-Cobb & Steptoe,
1998).
As part of the early surveillance review, each child receives a GA prior to bronchoscopy and
other procedures carried out in operating theatres as part of annual review, and this therefore may
be regarded as a peri-operative type experience. Whilst there is a plethora of research on peri-
operative experiences of children (e.g., Beringer, Segar, Pearson, Greamspet & Kilpatrick, 2014;
Chieng, Chan, Klainin-Yobas & He, 2014; Fortier & Kain, 2015; Lopez, et al., 2007), much less research
has been conducted on peri-operative experiences of parents whose children undergo anaesthesia.
Anxiety has been the most reported peri-operative emotional response for children,
reaching a prevalence of up to 60% (Kain, Caldwell-Andrews & Wang, 2002; Thompson, 1994; Wollin
et al, 2004). However, such figures do not exist for parents whose children undergo operative
procedures. Several randomised controlled studies assessing anxiety of parents have compared
parents who are present during the child’s anesthesia induction, compared to parents who are
absent from the procedure. Kain and colleagues (2000) and Bauchner and colleagues (1996) both
found that parents who were present for the child’s anaesthesia were less anxious than those who
did not attend. In contrast, Akinci, Kose, Ocal and Aypar (2008) found no differences between
parents who were present at their child’s anaesthetic induction compared to parents who were not
present. Therefore, debate continues as to the effectiveness of parent presence for both children
21 Parenting Children with Cystic Fibrosis
and parents. What is not yet clear is the experience of children and parents undergoing the annual
early surveillance procedures: the anaesthetic, bronchoscopy and post-anaesthetic recovery period;
and the associated psychological burden on parents’ ability to cope specifically with early
surveillance procedures, but also as a potential additional stressor to general CF care.
1.2.2.2 Parallels drawn from similar condition trajectories.
Research into parental experiences of life-threatening illnesses with similar treatment
management as CF may provide insight into how parents whose children undergo repeated early
disease surveillance might experience and cope with associated events. Caution should, however, be
exercised when generalising the experiences of parents of children with other chronic conditions
exposed to a variety of medical procedures that differ to those experienced by children undergoing
early surveillance for CF lung disease.
Cardiac
Cardiac conditions are often congenital and entail invasive procedures or surgical correction
in early life and may be life threatening or life shortening. These features parallel CF, and the early
surveillance program, to some extent. Considerable research of this condition has investigated
parental experiences and coping whilst their child is awaiting cardiac surgery. Similar to research on
parental experiences of GA, it has been reported that anxiety and emotional distress are elevated
prior to surgery, and that these emotions return to normative levels post-surgery (Brosig, et al.,
2007; Menahem, et al., 2008). To further understand experiences of parents whose children
undergo surgery, a qualitative study explored parental experiences and coping resources and found
that parents experienced a fear of their child dying that resulted in feelings of helplessness and
powerlessness (Salgado et al., 2011).
Cancer
Improvements in CF prognosis and treatments have followed a similar path to that of certain
paediatric cancers, such as leukaemia. It is possible to draw some similarities across surveillance and
treatment processes as well as parental psychological outcomes and coping skills from this
literature. Research has trended towards investigating resiliency factors in families, reporting
adaptability (Rosenberg, et al., 2014; Greeff, Vansteenwegen & Geldhof, 2014), communication
(Beek, Schappin, Gooskens, Huisman & Jongmans, 2014; Clarke, Sheppard & Eiser, 2008) and family
support (Nicholas, et al., 2009; Shortman, et al., 2013) as important components of a family’s
resiliency to a paediatric cancer diagnosis. Family resilience after a paediatric cancer diagnosis is a
recent construct receiving wide-spread interest (e.g., Van Schoors, Caes, Verhofstadt, Goubert &
Alderfer, 2015). However, understandably, most research into parental experiences of pediatric
cancer treatments has focused on negative psychological consequences, with depression, stress,
anxiety and post-traumatic stress syndrome (PTSS) and their relationships to adaptation receiving
22 Parenting Children with Cystic Fibrosis
the most attention (e.g., Greening & Stoppelbein, 2007; Hoekstra-Weebers, Jaspers, Kamps, & Klip,
2001; Kazak, Boeving, Alderfer, Hwang, & Reilly, 2005; Von Essen, Sjoden, & Mattsson, 2004).
The literature examining parental outcomes following their child’s diagnosis of paediatric
cancer suggests mothers are more negatively affected by their child’s cancer diagnosis and
subsequent treatment than fathers on several psychological outcomes. Mothers demonstrate more
depressive symptomotology (Von Essen, et al., 2004), more PTSS symptoms (Kazak, et al., 2005),
greater caregiver demand (Klassen, et al., 2010; Steele, Long, Reddy, Luhr & Phipps, 2003) and role
strain (Quittner et al., 1998), and a greater extent of perceived loss of control during required
treatments (Norberg & Boman, 2011). Norberg and Boman (2011) offered an explanation for these
differences. They suggested that generally mothers are in control of care and decisions about care,
and the parenting role may be more central to the identification of mothers than fathers. Therefore
mothers may be more affected by their child’s illness and subsequent treatment because of the shift
of the primary carer role from mother to HCPs.
Similar to research into parental experiences of CF, much less is known about fathers’
experiences of their child’s cancer diagnosis and subsequent treatment compared to the knowledge
of mothers’ experiences. Research has suggested that fathers with a university degree or higher
show greater anxiety about their child’s diagnosis than lower educated fathers (Mu, Ma, Hwang, &
Chao, 2002); fathers experience guilt related to role obligations due to their incapacity to provide
further care for their child because of their obligations to provide financial stability (Chesler & Parry,
2001); and fathers are more concerned about their family financial situation than family care (Hovey,
2005). It does appear that research into similar treatments, prognoses and disease trajectories may
provide some insight into parental experiences, and how parents cope with early surveillance for CF
lung disease. However these bodies of literature fail to take into account the exact nature of, and
processes involved in, early disease surveillance for CF lung disease. As there is currently no
published psychological literature examining the impact of early surveillance for young children and
infants with CF on parents, the literature review has attempted to provide evidence for the types of
coping resources and experiences that parents whose children undergo early surveillance may be
experiencing. Therefore, caution must be exercised when considering coping and psychological
outcomes of diseases that, whilst similar in terms of chronicity or severity or that have similar
treatment trajectories, may have distinct and important differences to CF.
The AREST CF program should be responsive to children’s, parents’ and families’
psychological well-being because of the potential burden of invasive surveillance procedures and
subsequent aggressive treatment. Therefore, extending knowledge about parents’ psychological and
social experiences associated both with their child’s CF and specifically with the early surveillance
process is a formidable task. It is likely to lead to adequate and evidence-based intervention
23 Parenting Children with Cystic Fibrosis
targeting identified issues and enhancing beneficial experiences and positive outcomes, which may
lead to better parent mental health outcomes and better child health and developmental outcomes.
Moreover, urgency exists for research to explore psychological and social factors that could
positively influence children’s, parents and families’ lives whilst maintaining the child’s physical
health through early surveillance of covert disease progression and subsequent treatment. Once this
is understood, education for medical staff and preventative or therapeutic intervention for parents
and/or children on these matters could play an important role in improving outcomes for parents,
children and their families.
Consequently, in an attempt to explore parenting within the context of early surveillance for
covert disease progression of a chronic condition, I recognised that the first step involved was asking
parents about their experiences, whether it be about diagnosis, treatment management, the early
surveillance program or other, broader, parenting experiences (Creswell, 2007). Given the paucity of
information about parents’ experiences within this unique context, a primary aim of my research is
to explore the lived experiences of parenting in the context of early surveillance for covert disease
progression. The particular aims of my research are outlined in detail in the next section.
1.2.3 Aims of the research
It is important to articulate the scope of my research. It was intended to neither advocate
for or against early surveillance for CF lung disease, nor to privilege early surveillance over other
clinical care pathways. The primary purpose of my research is to explore the lived experience of
parenting children undergoing early CF disease surveillance by providing a comprehensive account
of how parents construct their experiences and attribute meanings to early surveillance. Moreover,
is the aim to describe how parents cope with their experiences. Due to the unique experience of
parents in my research, the intention was not to generalise the experience of parenting within the
context of early surveillance for covert disease progression but rather to offer an in-depth
exploration of this phenomenon in its complexity and entirety against the wider contexts with which
it occurs. Whilst parents’ experiences of their child’s early surveillance were explored, I also
investigated the experience of parenting within the context of early surveillance.
1.2.4 Research questions
Given the research aims to explore; the lived experience and how parents construct their
experiences and attribute meanings to early surveillance for CF lung disease, and how parents cope
with their experiences, a qualitative inquiry paradigm is deemed methodologically appropriate.
Focussing on meaning of experiences meant that my research was ontologically consistent with
phenomenology (Guba & Lincoln, 1994), which emphasises the importance of investigating the lived
experience from the perspective of those who have experience with it (Barkway, 2001; Creswell,
24 Parenting Children with Cystic Fibrosis
2007; Liamputtong & Ezzy, 2005; Moutsakas, 1994; Patton, 1990) and focusing on how people
construct their experience in the context of their own life world (Creswell, 2007; Flood, 2010;
Patton, 2002; Zippel, 2010). Therefore, phenomenological research describes humanistic
phenomena as it is lived and experienced by people; rather than attempting to measure and
quantify an abstracted explanatory account (Crotty, 1996). A constructivist epistemology was used
to generate insights into how parents interpreted and formed meaning around their experiences.
Constructivism emphasises that construction of reality occurs as a dual process between two people
whereby it is constructed in the mind of the individual (Hansen, 2004) and co-constructed by
researcher and participant. An interviewing approach was used to collect data from parents, as it
was pragmatically and theoretically the most appropriate way to explore each for my research
questions (Creswell, 2007; Denzin & Lincoln, 2005; Riessman, 2008). To this end, research questions
in qualitative research paradigms are often open-ended and exploratory in nature (Elliot & Timulak,
2005). The over-arching research questions to address the aforementioned aims of my research are
as follows:
What is the lived experience of parenting in the context of early disease surveillance for CF?
What are parents’ experiences of early CF disease surveillance?
How do parents construct their experience and attribute meanings to early surveillance for
CF lung disease?
How do parents cope with the lived experience of parenting in the context of early
surveillance?
1.2.5 Implications of the research
My research contributes to a growing body of knowledge about the parenting experience in
paediatric chronic disease. Uniquely, findings from my research offer insight into the lived
experience of parenting a child undergoing early intervention and aggressive treatment for disease
progression. This type of information can assist in providing holistic support for parents and their
families. In practical terms, identification of psychological factors important in maintaining good
parent and family health, and insight into experiences of parents with children undergoing early
surveillance with AREST CF, may be used to inform existing and future policy decision-making and
models of best clinical practice towards promoting a positive parenting experience. Accordingly, my
research informs existing and future policy structures through identification of previous omissions,
misconceptions and anecdotal assumptions regarding parenting children in early surveillance and
aggressive treatment for their chronic condition. Moreover, my research contributes to
understandings of parenting children with chronic conditions as a process of acceptance and
redefinition involving positive appraisals and providing opportunities for resilience, rather than
25 Parenting Children with Cystic Fibrosis
focused solely on the adverse experiences and outcomes associated with paediatric chronic disease.
Additionally, the findings from my research highlight the relevance of supporting families from a
salutogenic approach rather than from the traditional deficit-focused models. Foremost, my findings
contribute to establishing a more holistic understanding of the parenting experience in paediatric
chronic disease and provide evidence that parenting a child with a chronic condition is a socially
constructed and therefore modifiable experience.
1.3 Overview of the thesis
The structure of my thesis has been organised into seven chapters. Each chapter begins with
an overview and concludes with a summary. Chapter one introduces the research topic by
identifying the need to understand the current experience and construction of parenting given the
current agenda of early surveillance and aggressive intervention for covert disease progression.
Moreover, is the need to understand parents’ attributions of meaning to their experiences and how
they cope with these experiences. With a dearth of information about the lived experience of
parenting in this context, I argue that to understand experiences of these parents, it needs to be
explored from the perspective of parents themselves. Additionally, recognising that theoretical
frameworks and clinical agendas are significant factors influencing our understanding of how parents
construct their experiences of, and attribute meanings to, their child’s chronic condition, as well as
how they cope, will provide the foundation of discussion in my thesis. The first chapter therefore
introduces the research context, development, focus and rationale of my research and outlines the
specific research aims and questions relevant to the exploration. This discussion leads to the next
chapter, which reviews current literature about parenting a child who has CF.
Chapter two critically examines relevant literature and context underpinning current
conceptualisations of parenting children with CF. As a focus of my research is on parents’
constructions of their experience, psychological perspectives of different aspects of parenting
children with CF, are of primary concern in review of the literature. Specifically, this chapter aims to
identify current understandings of how parents manage diagnosis and treatment within the first 7
years of life within modern approaches to CF management. This discussion then leads to the next
chapter, which reviews major theoretical developments in coping with parenting a child with a
chronic condition.
Chapter three outlines substantiations for theoretical and methodological choices made to
conduct the research. Firstly, this chapter aims to identify current theoretical understandings of
parenting a child with a chronic condition with a particular focus on theories focussing on coping,
adaptation and resiliency in the face of adversity. Accordingly, dominant theories and models
currently influencing contemporary debates and discussions on coping with parenting in paediatric
chronic disease are reviewed. Presented in this chapter is critical review on how parenting should be
26 Parenting Children with Cystic Fibrosis
considered in the context of broader family and community conditions including the role of
appraisals of the child’s condition and the family’s ability to cope with the demands, and their newly-
instituted patterns of family functioning to cope with the demands. Next is the rationale for
methodological choices of research design and paradigm. Specifically, this section aims to detail how
phenomenological inquiry, data collection and analysis, and sampling techniques informed the
conduct and output of the research. This leads to the next chapter, which outlines methodological
actions taken to conduct the research.
Chapter four describes the methodological approach used in my research. This chapter
initially discusses phenomenology and constructivist frameworks, which have informed and directed
my research conduct and output. As my research adopted a qualitative methodology to explore
parents’ experiences, constructions, and attributions, the philosophical underpinnings of a
qualitative paradigm are detailed. Participant characteristics, research procedures, ethical
considerations, my role as a researcher, and data collection and analysis techniques are presented.
Therefore, the aim of this chapter is to outline the research methodology and the means by which
the research was conducted, and findings from the data were derived and substantiated. The next
chapter presents the findings and interpretations of the data.
Chapter five introduces the research findings relevant to addressing the research questions.
Specifically, this chapter presents findings on parents’ experiences and constructions of parenting in
the context of early surveillance for CF lung disease. Moreover, the chapter presents findings about
how parents cope with their experiences, and how they attribute meaning to early surveillance for
CF lung disease. Similarities and points of difference are reviewed, with findings presented in a
structure of five major themes and their associated sub-themes. These major themes included:
redefined expectations and reimagined identity; redefined reality (consisting of sub-themes -
collapsed and redefined expectations and new normal); understanding the unknown and
understanding uncertainty (consisting of sub-themes – understanding CF and understanding early
surveillance for CF lung disease); good days and bad days; fluctuation between positive and negative
outlook on life (consisting of sub-themes – bad days, good days and fluctuation between good days
and bad days); early surveillance is a significant event (consisting of sub-themes - indicator of
treatment efforts and examination of parenting competence, parental outcomes directly from early
surveillance and fear from diagnosis exacerbated at each annual review can result in anxiety); and
early surveillance is in the best interest of my child (consisting of sub-themes - perceived clinical
benefit to child health and cognitive dissonance results in anxiety and/or ambivalence). The findings
are critically examined with reference to current understandings and constructions of parenting in
paediatric chronic disease. Theoretical application of the findings is addressed in the next chapter.
27 Parenting Children with Cystic Fibrosis
Chapter six provides further discussion of the findings by presenting a critical consideration
of how relevant research findings can be applied to theoretical models presented in chapter three,
with particular reference to how coping and adaptation can be achieved from a family perspective.
Parental characteristics and parenting processes that contribute to family adaptation are identified,
along with how some factors that may contribute to adaptation can be negatively influenced by the
early surveillance experience. The chapter concludes by proposing that strength-based, salutogenic
models of care and support are appropriate conceptualisations of parenting children with chronic
conditions who undergo early surveillance for covert disease, and appear better approaches than
the traditional deficit-models of care and support. Moreover, resiliency as an outcome for families
facing adversity should be of primary consideration in current models of care for the entire family.
Chapter seven is the concluding chapter of the thesis that pulls together a summary of the
salient findings and issues raised across the research. Within context of the research questions, my
findings are discussed in relation to the implications they have for policy and clinical practice, and
theoretical development of parenting in paediatric chronic disease. The limitations of my research
are then discussed with particular reference to opportunities and recommendations for future
research and practice before concluding with a post-script anecdote about my research journey.
28 Parenting Children with Cystic Fibrosis
CHAPTER 2
Cystic fibrosis in the 21st century
This chapter describes cystic fibrosis (CF) and contextualises paediatric CF in an
Australian context. The chapter is divided into four sections, each of which contributes
to the rationale for conducting the research within the chosen methodology. In the first
section, CF will be comprehensively explained including current diagnosis practices and
treatment options. Living with CF as a child will be outlined before presenting the
burden of CF management experienced by parents and their families. The second
section will review parenting in paediatric CF by reviewing empirical evidence of how
parents adjust to, construct meaning of, and experience, their child’s CF diagnosis and
parenting a child with CF in their first 7 years of life. This section will conclude with
discussion about contribution of parental characteristics on child mental and physical
health outcomes. The third section outlines empirical support for parents’ strategies
and abilities for coping with a child’s CF management, before viewing how parents cope
from a resiliency perspective. The fourth section of the chapter provides specific
rationale for conducting the research, culminating in its aims and significance.
29 Parenting Children with Cystic Fibrosis
Out of your vulnerabilities will come your strength…Sigmund Freud
2.1 What is CF?
2.1.1 Prevalence, life expectancy, causes and clinical features.
Cystic fibrosis (CF), the most common life-shortening autosomal recessive disease of
Caucasian people, is a multi-systemic and genetically inherited disease affecting 1:2,500 - 3,500 live
born infants (Daly, et al., 2007; Davies, et al., 2007; Montgomery & Howenstine, 2009) though its
frequency varies in specific groups (Daly, et al., 2007). An analysis of literature reviews, surveys, and
registry data of European Union (EU) countries revealed mean prevalence rates of 0.737:10,000 live
born infants in the 27 EU countries, which is similar to the value of 0.797:10,000 live born infants in
the United States, with one outlier, namely the Republic of Ireland at 2.98:10,000 live born infants
(Farrell, 2008). The high Irish prevalence rate is likely attributable to a high genetic mutation
prevalence and consanguinity (Farrell, et al., 2007). Current Australian prevalence rates are
approximately 1:3,000 live births (Australian Bureau of Statistics, 2009; Cystic Fibrosis Australia,
2013).
Progress in understanding CF and its impact on disease management has been rapid over
the past 20 years, with CF changing from a fatal childhood digestive and lung disease to a complex
multi-systemic disease extending into adulthood (Davies, et al., 2007). With advances in science,
medicine and technology, life expectancy of CF patients has increased dramatically over the past
four decades, increasing from less than 1 year in 1960 to a median survival age of approximately 40
years now (UK Cystic Fibrosis Registry, 2012). Availability of direct mutation analysis and the gene
sequence were turning points in the history of CF and opened a new era of research into molecular
and cellular studies in CF research (Zielenski, 2000). This has resulted in identification of CF gene
mutations, which has allowed early diagnosis and therefore early and prophylactic treatments for
CF, culminating in increasing life expectancy for people living with CF.
Cystic fibrosis is caused by mutations identified on the long arm of chromosome 7 (7q.31.2),
and identification of the mutation was first isolated by positional cloning in 1989 (Kerem, et al.,
2005; Riordan, et al., 1989; Rommens, et al., 1989). The mutation occurs in what is known as the
cystic fibrosis transmembrane conductance regulator (CFTR) gene (Riordan, et al., 1989). Different
mutations in the CFTR gene result in varying phenotypes of the disease (Rowntree & Harris, 2003).
The same mutation may also result in various phenotypes, for example, two people with the same
mutation may or may not be pancreatic sufficient (Davies, et al., 2007). Expositions for this anomaly
are currently being proposed in the literature, with environmental (Folkesson et al., 2012) and
chromosomal interactions (Drumm, Ziady, & Davis, 2012) put forth as the most plausible
explanations.
30 Parenting Children with Cystic Fibrosis
Cystic fibrosis primarily affects chloride channel transport in epithelial cells in the intestine,
respiratory system, pancreas, gall bladder and sweat glands (Rowntree & Harris, 2003); therefore
manifesting in a number of organs, most notably in the upper and lower airways, pancreas, bowel,
and reproductive tracts (Davis, 2006) leading to chronic obstructive lung disease, exocrine pancreatic
insufficiency, elevated chloride concentration and infertility in males due to absence or obstruction
of the vans deferens. Females also suffer from reduced fertility, possibly due to altered cervical
mucus properties (Daly, et al., 2007; Zielenski, 2000). The lack of CFTR function causes
gastrointestinal issues because the intestines are unable to break down fats (John Hopkins Cystic
Fibrosis Center, 2015), requiring continue enzyme replacement therapy to help the intestine absorb
fat-soluble vitamins. Though for most patients with CF, lung disease is the most important issue in
terms of symptoms and treatment because it is the most likely cause of death (Davies, et al., 2007).
The sweat gland duct is also affected; concentration of salt in sweat of CF patients is elevated (3-5
times more than normal levels) (Daly, et al., 2007). The identification of elevated salt in sweat is a
clinical indication that CF is present, which is usually conducted after an abnormal Guthrie heel-prick
test (Guthrie & Susi, 1963). The most recognised symptom of CF disease is chronic obstructive lung
disease (Montgomery & Howenstine, 2009). Chronic, suppurative lung disease causes over 90% of
the morbidity and mortality associated with CF, leading to premature death from respiratory failure
in early adulthood (Rajan & Saiman, 2002).
2.1.2 Chronology of CF into the 21st century.
Based on historical medical documents, CF apparently appeared about 3,000 years ago,
possibly due to migration of peoples, gene mutations, and new conditions in nourishment (Busch,
1990). The complete clinical range of CF symptoms was not recognised until the 1930s (Andersen,
1938), however aspects of CF were identified much earlier. For example, in the 18th century,
Northern European folklore warned, translated to English, woe to that child which when kissed on
the forehead tastes salty. He is bewitched and soon must die (Busch, 1990). The significance of
saltiness was later demonstrated through the association between excessive salt content in sweat of
affected children and the condition (Di Sant’Agnese, Darling, Perera & Shea, 1953), which resulted in
the measurement of chloride and sodium in sweat as the diagnostic standard for the condition
(Gibson & Cooke, 1959).
Aspects of CF were reported in the medical literature throughout the early 1900s, including
a correlation between coeliac disease, CF of the pancreas and bronchiectasis (Fanconi, Uehlinger, &
Knauer, 1936). In 1938 Dorothy Hansine Andersen first described CF using the term cystic fibrosis as
a reference to the characteristic scarring, or fibrosis, and cyst formation within the pancreas. This
was the result of many electrophysiological studies highly suggestive of a defect in chloride transport
of CF secretory epithelial cells (Quinton, 1983). Andersen was the first to describe characteristics of
31 Parenting Children with Cystic Fibrosis
CF of the pancreas and to associate it with intestinal and lung disease prominent in CF. She also first
hypothesised that CF was a recessive condition, and she led the use of pancreatic enzyme
replacement to treat affected children. Subsequent research has found more than 1,000 different
mutations that cause CF, for example, one study alone found more than 50 mutations in a sample of
350 German patients with CF (Dörk et al., 1994), with another study finding a further 17 mutations in
a sample of 137 French CF patients (Claustres et al., 1993). Since the discovery of countless CF-
causing gene mutations, many studies have investigated the correlation between CFTR genotype
(that is, a person’s genetically coded information) and CF disease phenotype (that is, the physical
manifestation of genetically coded information) (Montgomery & Howenstine, 2009; Rowntree &
Harris, 2003; Zielenski, 2000).
Therefore, early understandings of CF disease and its effect on different body organs has
advanced to contemporary understandings of CF mutations, their molecular consequences, their
disease mechanisms, and environmental and CF gene modifiers. This has resulted in CF being
classified as a disease with a complex, multi-faceted clinical phenotype that is the sum of a number
of variable clinical components that arise from the underlying genotype in a unique and organ-
specific manner. Lastly, studies in CF have illustrated that CF genotype-phenotype can be
confounded by secondary genetic and environmental factors and not only by different molecular
mechanisms of underlying mutations (Montgomery & Howenstine, 2009; Rowntree & Harris, 2003;
Zielenski, 2000).
Due to the complexity in CF gene dysfunction and variability in genotype-phenotype
outcomes, hope for a cure in the foreseeable future is now understood as not as realistic as it was
when the CF gene was discovered back in 1989. As an example, attempts to insert normal genes into
epithelial cells have not been as therapeutically useful as anticipated (Knowles, et al., 1995). With
this understanding, researchers are now focussing on gene therapies as a way to manipulate the
dysfunction caused by the genetic mutations. Kalydeco (Ivacaftor) is one such treatment that has
shown promising results for individuals with the G511D mutation (Ledford, 2012). Unfortunately for
infants and young children with CF, Kalydeco is only currently registered for individuals with CF who
are 6 years of age and older.
2.1.3 CF diagnosis: Current Australian practice via newborn screening.
The most discriminatory diagnostic test for CF is the measurement of sweat electrolyte
levels (Di Sant’Agnese, et al., 1953), and it remains the gold standard for diagnosis (Montgomery &
Howenstine, 2009; Rosenstein & Cutting, 1998). The median age for diagnosis with classic CF is
approximately 6 months, whilst diagnosis of non-classic forms of the disease with milder symptoms
can be delayed. Elevated levels of a particular protein, immunoreactive trypsinogen in the blood
(Crossley, Elliott and Smith, 1979), is the most common phenotypic attribute utilised to establish a
32 Parenting Children with Cystic Fibrosis
definitive CF diagnosis, and was implemented as part of newborn screening (NBS) in New Zealand
and regions of Australia in 1981 (Barben, et al., 2012). In Australia, NBS is conducted through a
national public health program.
As a public health program, NBS is designed to screen infants for a number of conditions
that are not clinically evident during the newborn phase, with screening usually conducted shortly
after birth, usually 1-3 days (Lindner, et al., 2011). Generally conducted by state or national
governing bodies, NBS programs aim to screen all infants born within their jurisdiction, with the
number of conditions screened for decided by each governing body (Loeber, et al., 2012). Whilst
there is some variation between states and territories as to which conditions are screened for in
Australia, the following conditions are currently screened for nation-wide; phenylketonuria,
galactosaemia, primary congenital hypothyroidism, some rare metabolic conditions and CF (Barlow-
Stewart, Emery & Metcalfe, 2007). If left undiagnosed, many of these conditions can cause
permanent physical and intellectual disability.
The implementation of NBS internationally and the development of AREST CF has resulted in
prophylactic treatment for children and infants with CF in an Australian context, and a paradigm shift
from reactive treatment to proactive early-life disease surveillance and therapeutic intervention.
Much of the physiological improvement in long term health outcomes has been attributed to
improvement in diagnosis and therapy. Therapeutic interventions administered before the onset of
symptoms, such as those offered by AREST CF, may have the greatest long term benefit (Farrell, et
al., 2001). This has led to the notion that asymptomatic diagnosis and early treatment may optimise
early nutritional status and prevent or reduce irreversible pulmonary damage, thereby improving
outcome and quality of life in people living with CF (Southern et al., 2007).
Although NBS now occurs in Australia and several countries across the world, its protocol
and subsequent treatment following diagnosis is not systematically conducted, with varying
treatment protocols across countries. For example, some paediatric CF centres administer treatment
following positive results for inflammatory markers from bronchoscopies (e.g., Princess Margaret
Hospital and Royal Children’s Hospital in Australia, and the Great Ormond Street Hospital in
England), whereas other CF centres do not conduct routine bronchoscopies in young children. The
various treatment following diagnosis means that a systematic CF treatment approach does not
currently exist. This indicates a need for a standardised, and empirically supported, treatment
protocol that serves to improve clinical outcomes and quality of life for children with CF.
2.1.4 What is it like to be a young child with CF in the 21st century?
Today, children with CF live relatively conventional lifestyles, albeit with some family
adjustments requiring parents to complete daily treatment regimens of physiotherapy and enzyme
replacement therapies. Currently, approximately 70% of children with CF are diagnosed before the
33 Parenting Children with Cystic Fibrosis
age of 1 year (Walters & Mehta, 2007), with clinical symptoms often present at birth (FitzSimmons,
1993). Respiratory symptoms are present in more than 50% of newborns, with infections such as
Pseudomonas aeruginosa occurring in early infancy (Walters & Mehta, 2007) and pancreatic
abnormalities occurring within days of birth (Littlewood, 2007). As children get older, they may
experience further CF-related symptoms and also pain associated with the disease including
headaches, muscoskeletal pain and abdominal cramping (Koh, Harrison, Palermo, Turner, &
McGraw, 2005; Palermo, Harrison, & Koh, 2006). These symptoms may also impact upon sleep
(Quittner, Barker, Marciel & Grimley, 2009), which brings with it a multitude of both psychological
and physiological issues that can be affected. For example, research has shown that sleep can
adversely affect emotional responses, behaviour, attention and academic performance in children
(Berger, Miller, Seifer, Cares, & Lebourgeois, 2012; Fallone, Acebo, Arnedt, Seifer, & Carskadon,
2001; Gozal, 1998; Yue, Conrad, & Dimsdale, 2008). Therefore, children’s quality of life may also be
detrimentally affected by the physical symptoms caused by CF (Palermo, et al., 2006).
Improvements in life expectancy of CF patients has also shifted the focus of CF research and
clinical care from improving physiological outcomes toward the complex interplay among
behavioural, psychosocial and health outcomes (Barker & Quittner, 2010). This change in focus has
been marked by a growing number of patient-reported outcomes, such as health-related quality of
life and efforts to address behavioural and psychosocial challenges, such as adherence and
depression (Quittner, Modi, Lemanek, Ievers-Landis & Rapoff, 2008; Quittner, et al., 2008). However
for ethical and practical reasons, this shift has not tended to focus on very young children with CF as
much as adolescents and adults with CF.
As children with CF experience frequent hospital visits and repeated medical procedures
(Thompson & Gustafson, 1996), it is thought that this can contribute to onset of psychological and
behavioural difficulties (Boyd & Hunsberger, 1998; Pao & Bosk, 2011; Thompson, Gustafson, Hamlett
& Spock, 1992a). Häglöff (1999) suggested that children of various ages were at higher risk of
psychological problems following increased follow-up surgeries from early interventions. Another
area of concern is children’s reactions to painful or distressing medical procedures (Hedström,
Haglund, Skolin, & von Essen, 2003). Children continue to find having a needle as one of the most
distressing aspects of attending hospital (Duff & Oxley, 2007; Ellis, Sharp, Newhook, & Cohen, 2004;
Kennedy, et al., 2008). One study in CF found over 30% of children felt extremely or very frightened
of venepuncture, especially prior to needle insertion (Duff & Brownlee, 1999). Together, these
studies highlight environmental factors which may interact with distressing aspects of CF disease.
Moreover, the majority of these studies did not differentiate across all age groups, therefore failing
to identify at what age such psychological and behavioural difficulties may begin to manifest. By
further identifying age of onset for such difficulties, or which personality characteristics may
34 Parenting Children with Cystic Fibrosis
contribute, clinicians and other allied health professionals would be better-equipped to implement
strategies to combat psychological and behavioural difficulties that occur as a result of medical
procedures children with CF must undergo. There is also potential for implementation of targeted
interventions for prevention rather than therapy if research can pinpoint age groups for which such
difficulties may begin to manifest.
2.1.5 Burden of early CF care on families in the 21st century.
Improvements in CF management and health outcomes have meant that the landscape of
parents’ experiences of CF has changed. As CF is no longer classed as a fatal childhood condition,
previous experiences of fatality in childhood and morbidity from the condition generally bear less
relevance today for parents. However, due to advancements in treatment, CF care is now primarily
the responsibility of parents. Consequently, despite improvements in life expectancy, CF can still
impose significant burden on children, their parents and their families. In particular, the shift in
responsibility to home treatment and daily therapy can cause treatment burden for parents
(Bregnballe, Schiotz, Boisen, Pressler & Thastum, 2011; Quittner, Drotar & Ievers-Landis, 2000; Ziain,
et al., 2006). Moreover, for parents whose children undergo early surveillance of lung disease,
knowledge of how their child’s disease is developing in their early years of life may cause a
psychological burden for parents (Sawicki, et al., 2015).
Parents generally establish a family routine that is centered around CF care and
management (Agent & Madge, 2007). Medical activities such as performing routine chest
physiotherapy or administering oral medication to an infant may feel unnatural and awkward to
parents, and the parenting role may become confused with a more medicalised role during these
early years (Agent & Madge, 2007). The performance of daily treatments is also time consuming, and
can interfere with parents’ social functioning and work roles (Quittner, et al., 1998). Managing
treatment increases parental stress and has been associated with depression, poor sleep quality and
marital strain in parents of young children with CF (Glasscoe, Lancaster, Smyth, & Hill, 2007;
Quittner, et al., 1992; Yilmaz et al., 2008). This means that successful management of the child with
CF depends largely on the ability of the family to change its lifestyle. Families must work together to
accommodate the time-consuming demands of a complicated daily treatment regimen consisting of
a multitude of therapies, according to the recommendations of the healthcare team (McCubbin, et
al., 1984).
Psychosocial factors that function as moderators of treatment adherence in parents have
been a dominant research focus for a number of reasons. These include the responsibility of CF care
within the home, the high treatment burden associated with CF management, and the recognised
importance of treatment adherence for the long-term management of adolescent and adult CF.
Estimates of treatment adherence by parents vary according to method of measurement, type of
35 Parenting Children with Cystic Fibrosis
treatment and age. In one of the few studies measuring adherence to several treatment
components using a number of assessment methods, Modi and colleagues (2006) reported
adherence rates for children ranging from 22% to 71% for objective measures and 67% to 100% for
self-reported measures, suggesting inflated adherence rates for self-reported measures. The study
also found significant variability between treatments, a finding that is consistent with other CF
studies (Quittner, et al., 2000; Quittner, Espelage, Ievers-Landis & Drotar, 2000; Quittner, et al.,
2008). The literature suggests that treatment adherence, and more so the reporting of it, is a
complex matter.
Maternal depression has been associated with low adherence to airway clearance (Smith,
Modi, Quittner & Wood, 2010), and identified as an important predictor of poor enzyme adherence
and shows short-term effects on weight gain and growth in children (Quittner, Barker, Geller, Butt &
Gondor, 2007). Another study found significant associations between parental stress, depression,
eating and sleeping difficulties in children on the one hand, and poor adherence to nebulised
medications during the preschool years on the other (Ward, Massie, & Glazner, 2009). Sheehan and
colleagues (2014) reported correlations between parental avoidant coping and internalised behavior
problems arising in children, as well as child mealtime/eating difficulties, which can result in a low
nutritional status for children with CF. Together, these findings indicate that parental psychological
issues such as depression and stress are likely to have an adverse effect on treatment management
(e.g., attending clinic appointments) and adherence (e.g., maintaining recommended home
treatments). Therefore, annual screening of depression and other potential psychological issues and
adequate support for parents of children with CF during the child’s early years may improve parental
and child health outcomes. This underscores the notion that parents are the most important
managers of treatment for young children with CF, and will require continuous family, medical and
social support in their roles.
Families of older children with CF have been shown to function better than families of
younger children with CF (Knafl, et al., 2013), and better family functioning has been associated with
better treatment adherence (Everhart, Fiese, Smyth, Borschuk & Anbar, 2014; Patterson, et al.,
1993). Collectively, studies to date highlight the importance of parent well-being and family
functioning in treatment adherence. Furthermore, children in these studies would have been old
enough to administer their own medication. Family functioning and treatment adherence has not
been sufficiently addressed in families who have an infant or young child with CF. It is important to
understand how, why, and at what point parent well-being and family functioning influences
treatment adherence. An additional implication of investigating treatment adherence in families
across large, undifferentiated age ranges is the difficulty in determining the relevance of findings to
families with very young children. Treatment management and adherence in the older population
36 Parenting Children with Cystic Fibrosis
primarily focusses on autonomous disease management and not that of parent-led management,
which is the case for the younger population.
The hidden, or asymptomatic, nature of early CF disease progression has dominated
research in the young population, attention has not therefore been given to parents’ experiences of,
and reactions to, how a child’s disease is developing. Nor has research focussed on how parents
cope with such knowledge. Though it is now understood that covert disease progression can start
very early in life (Ramsey, et al., 2016; Stick, et al., 2009), psychological research into parental
understandings of such information are yet to be integrated.
2.1.6 Summary and conclusion.
Cystic fibrosis is the most common life-shortening disorder of Caucasians, with systemic
involvement including the lungs and pancreas. Recent advances in NBS have provided opportunity
for early surveillance and proactive, aggressive treatment of CF. Life expectancy of children born
with CF has increased exponentially over the past 30 years, though both physiological and
psychological issues remain important issues for children and their families. Moreover, parent
mental health and family functioning can be affected due to home treatment and daily therapy.
Currently, clinical guidelines and standards of care are generally limited to medical aspects of CF
with less consideration of accompanying psychosocial aspects. Although national standards of care
(Bell & Robinson, 2008) advocate for a psychologist and social worker as part of the multidisciplinary
CF team, national consensus on how to assess psychological health of parents and families and how
to address these issues within clinical care is urgently needed. However, a recent consensus
statement from the International Committee on Mental Health in Cystic Fibrosis asserted the need
to assess anxiety and depression annually in parents and caregivers of children with CF (Quittner, et
al., 2015).
2.2 Families living with CF
2.2.1 Psychological adjustment to a CF diagnosis.
A CF diagnosis in a newborn is undoubtedly devastating news for parents. For most parents
the diagnosis of CF in their newborn infant is unexpected (Massie, Forbes, Dusart, Bankier, &
Delatycki, 2007) and often parents have little knowledge about the disease or carry out-dated
misconceptions about its lethality in childhood (Jedlicka-Köhler, Götz, & Eichler, 1996) despite pre-
diagnostic genetic counselling during NBS (Tluczek, Orland, Nick, & Brown, 2009). The psychological
impact of a CF diagnosis is well documented in the literature with disruption of family routine, shock,
denial, anger, guilt, worry and sadness ensuing (Eiser, Zoritch, Hiller, Havermans & Billig, 1995;
Gayton, Friedman, Tavormina & Tucker, 1977; Marvin & Pianta, 1996; Priddis, Dunwoodie, Balding &
Douglas, 2010; Quittner, et al., 1992; Turk, 1964; Venters, 1981). A crisis period is generally reported
to last up to one year (Venters, 1981), with a number of studies reporting that, after a period of
37 Parenting Children with Cystic Fibrosis
initial disruption and adjustment, families generally return to relatively common and conventional
levels of family functioning (Cowen, et al., 1985; Cowen, et al., 1986; Knafl & Gilliss, 2002; Pfeffer,
Pfeffer, & Hodson, 2003; Sawyer, 1992; Szyndler, et al., 2005).
However, the above-mentioned research is contrasted with the notion that following
diagnosis, parents continually experience negative psychological outcomes compared to controls
and norms, such as more depressive symptoms, higher stress and anxiety, and greater role strain
(Blair, Cull, & Freeman, 1994; Breslau, Staruch, & Mortimer, 1982; Brucefors, Hochwälder, Sjövall, &
Hjelte, 2015; Goldberg, Morris, Simmons, Fowler, & Levison, 1990; Mullins et al., 1991; Quittner, et
al., 1992). As a composite of these arguments for parental adjustment to diagnosis, Duff and
Brownlee (2008) posited that once diagnosis is confirmed, parents typically continue to experience
fluctuating states of psychological distress and normalcy as they take on the role of medical parent.
Phenomenological studies into the experience of caring for a child with CF confirm this supposition.
Parents appeared to be on a continuous journey of uncertainty as they struggled to adapt to their
child’s diagnosis, alternating between periods of adjustment, facing adversity and readjustment due
to the progressive nature of the disease and changing health outcomes of their child (Glasscoe &
Smith, 2008, 2011; Jessup & Parkinson, 2010). From these studies, families generally follow a pattern
of initial crisis following diagnosis replaced somewhat later by adaptation and acceptance, with
cyclical periods of adversity and re-adjustment possibly associated with developmental milestones
and/or disease progression.
2.2.2 Outcomes of parenting a child with CF.
Similar to parenting a healthy child, parenting a child with CF can bring both hardships and
joy to parents. Theoretical approaches to understand parenting a chronically ill child traditionally
stem from a psychopathologic perceptive. Whether parental outcomes in paediatric CF are a product
of these early theoretical underpinnings is not the topic of debate here; the purpose of discussion
here is to provide a substantial summary and critique of the literature, and what is and is not
reported in terms of parents’ experiences and outcomes. Notwithstanding the trend towards
investigating psychopathologic outcomes, parental outcomes in paediatric CF include stress,
depression and anxiety, guilt and chronic sorrow, with some reported effects on parental identity
formation and parenting self-efficacy.
2.2.2.1 Parental stress, depression and anxiety.
The life-shortening nature of CF, time consuming treatments and ongoing morbidity (Davies,
Alton, & Bush, 2007) place pressure on parents beyond normal parenting duties. With the diagnosis
of CF generally occurring in the first year of life, parents must not only learn about their new baby,
but also about a new and often frightening condition requiring rapid change and adjustment in
parenting roles and plans for the future (Gotz & Gotz, 2000; Quittner et al., 1998; Quittner, et al.,
38 Parenting Children with Cystic Fibrosis
1992). High levels of continual parenting stress and depression are often reported (Quittner et al.,
2008; 2014).
In terms of psychological effects of CF, early studies presumed that children with CF and
their families would experience psychopathology, which was predictably found (Lawler, Nakielny &
Wright, 1966; Pinkerton, 1969; Teicher, 1969; Tropauer, Franz, & Dilgard, 1970; Turk, 1964). Since
the 1960s, most research into psychological effects of CF has focused on maternal reactions and
responses. Such research has generally shown that mothers experience negative effects of CF, such
as heightened depression and anxiety (Mullins, et al., 1991; Quittner, et al., 1992; Yilmaz, et al.,
2008), though high rates of depression are reported for both mothers and fathers soon after
diagnosis (Glasscoe, et al., 2007). A current international epidemiological study is underway,
researching population-based estimates of depression and anxiety, and examining their associations
with health outcomes (Quittner, et al., 2008).
This large-scale ongoing epidemiological study (The International Depression/Anxiety
Epidemiological Study – TIDES), that commenced in 2007, is measuring levels of anxiety and
depression in both patients with CF from ages 12 – 35 years and caregivers of children with CF ages 1
- 17 years in 18 countries (http//:www.tides-cf.org/). An early study from this collaboration reported
that almost a quarter (20%) of caregivers reported clinical levels of depressive symptomatology, and
more than a quarter (28%) reported elevated levels of anxiety. What this study and other studies
from Germany and America within the TIDES collaboration (Besier, et al., 2011; Driscoll, et al., 2009)
indicate is that caregivers adapt surprisingly well to the demands of their infant’s or young child’s
condition. However, these studies also indicate that a substantial proportion of caregivers report
elevated levels of psychological distress associated with caregiving for a child with CF. The TIDES
study will go some distance in consistently determining prevalence and severity of anxiety and
depression in patients and their caregivers across countries and across cultures.
However, critiques of the TIDES study thus far have reported methodological weaknesses
due to inconsistencies in psychometric measurement across countries (Webb & Bryon, 2014),
bringing into question clinical significance of reported findings. Additionally, whilst the design of the
study enables risk factors for depression to be identified, which will help better our understanding of
the nature of psychological needs of parents with children who have CF, such knowledge of
increased risk for parental depression in paediatric chronic disease is not new (Besier, et al., 2011;
Glasscoe, et al., 2007; Goldbeck, et al., 2008).
Research has also shown that mothers’ experiences of CF are more negative than fathers,
with mothers experiencing more stress (Van Os et al., 1985) anda greater incidence of depression
than fathers (Quittner, et al., 2014). However, fathers of children with CF had more depression
compared with fathers of healthy children (Brucefors, et al., 2015; Goldberg, et al., 1990). To date,
39 Parenting Children with Cystic Fibrosis
there has been much less research on fathers’ experiences of their child’s CF, with suggestions in the
literature that fathers experience greater financial strain and have less time to spend with their
children and family as a result of these financial demands (Blair, et al., 1994; Goble, 2004; Hayes &
Savage, 2008; Nagy & Ungerer, 1990). To determine what factors lead to parental stress and
depression for both parents, and what leads to higher parental stress and depression in mothers,
processes that lead to family stress in relation to their child’s diagnosis have been researched.
Quittner and colleagues (1992) found that mothers often took primary responsibility for their child’s
treatment, which led to increased stress and more depressive symptomatology.
Beyond the diagnosis phase, few studies have assessed psychological functioning of parents
(Quittner, et al., 2009). Consequently, general trends of psychological functioning and adaptation for
parents of infants and young children with CF have not yet been established. Psychological
adaptation to a child’s CF has been likened to the bereavement process, with distinct stages of
shock, belief, anger, adjustment and adaptation (Cousino & Hazen, 2013). This does not, however,
accurately reflect commonly reported experiences by parents. Despite hope of gene therapy and
increased longevity of life, parents continue to worry they will outlive their child. What seems more
appropriate is the notion of chronic sorrow, with dynamic phases of normalcy and routine,
interspersed with intense sadness and mourning (Duff & Oxley, 2007).
2.2.2.2 Guilt and chronic sorrow.
Due to the devastating nature of paediatric chronic disease, guilt and chronic sorrow are
commonly-reported outcomes for parents of children with both chronic and life-limiting conditions
including type 1 diabetes, asthma, cerebral palsy, neurological disability and leukaemia (Bowes, et
al., 2009; Cashin, et al., 2008; Johnson, 2000; Masterton, 2010, Weng, et al., 2012; Whittingham, et
al., 2013; Zhou, Yi, Zhang & Wang, 2014). This is also the case for autosomal recessive conditions
whereby hereditary is the mechanism for disease etiology including sickle cell disease,
phenylketonuria, Sanfilippo syndrome and thalassemia (Grant, et al., 2013; Weng, et al., 2012). Guilt
about the hereditary component of CF is common (Tluczek, Koscik, Farrell & Rock, 2005), and is a
well-established finding of parental psychological effects of a CF diagnosis in an infant (Hodgkinson
& Lester, 2002; James, Hadley, Holtzman & Winkelstein, 2006; Jedlicka-Köhler, et al., 1996; McCraea,
Culla, Burton & Dodge, 1973; Tropauer, et al., 1970). Feelings of guilt in parents of children
diagnosed with CF have been shown to adversely affect parenting self-esteem (Courlet, Dodane &
Garnier, 2014) and development of competent parental identity (Hodgkinson & Lester, 2002), which
can then contribute to adverse parenting behaviours (Teicher, 1969) and hostile parent-child
relationships (Tropauer, et al., 1970). Similarly, parents experiencing guilt for their child’s diagnosis
in other paediatric chronic conditions has been found (Grant, et al., 2013; Weng, et al., 2012; Zhou,
et al., 2014).
40 Parenting Children with Cystic Fibrosis
Though understood as common amongst parents of children with chronic conditions and
disabilities (Bowes, et al., 2009; Damrosch & Perry, 1989; Masterton, 2010, Neilsen, 2013; Nikfarid,
Rassouli, Borimnejad & Alavimajd, 2015; Paul, Sheilini & Roach, 2014; Patrick, 2014; Whittingham, et
al., 2013), prevalence rates as well as possible effects of chronic sorrow on parents of children with
CF and their families have not been sufficiently studied (Moola, 2012; Priddis, et al., 2010;
Shumaker, 1994). Referred to in the trauma literature as a sense of recurring, extended and
profound sadness (Gravelle, 1997; Olshansky, 1962), chronic sorrow generally occurs because of the
profound sense of loss parents feel as a result of their child’s diagnosis including; loss of traditional
parenting functions and role, loss of the ideal child and family, and loss that the child will experience
(Fernández-Alcántara, et al., 2015; Cashin, et al., 2008; Whittingham, et al., 2013). Effects of chronic
sorrow on parents of children with CF have not been as extensively studied as effects of guilt on
parents, however Shumaker’s (1994) doctoral research specifically explored outcomes of chronic
sorrow for these mothers. She found high rates of depression and anxiety for mothers experiencing
chronic sorrow. More recently, both Priddis et al. (2010) and Moola (2012) made reference to
comparable adverse effects of chronic sorrow for parents of children with CF. Therefore, as the
landscape of CF diagnosis and treatment continue to evolve, so should the focus of research follow
in its footsteps. An example of such a transition could be to understand if the nature of chronic
sorrow changes with the nature of earlier diagnosis, improved health outcomes and knowledge of
early disease progression.
A concept analysis of the defining characteristics of chronic sorrow by Eakes, Burke and
Hainsworth in 1998 resulted in a context-specific theory of chronic sorrow for parents experiencing
paediatric chronic conditions. The theoretical model was developed through interviews with parents
of chronically ill children. It describes critical times when parents are likely to experience episodes of
chronic sorrow by identifying antecedents and trigger events. The model further describes
management methods by the person experiencing chronic sorrow and by those offered for
therapeutic purposes. Clements, Copeland and Loftus (1989) and Shumaker (1994) qualitatively
explored these critical time points in families of children with CF, and found that chronic sorrow was
present at all developmental stages and disease-related time points including exacerbations of CF,
hospital admission and independence in adolescence. These times brought about recurring and
cyclical periods of chronic sorrow for parents. Predicting these critical times enables the health care
team to optimise and maximise the effectiveness of treatment based on the conceptual model of
chronic sorrow by Eakes and colleagues.
Whilst these studies are dated, they represent the small number of studies paying particular
attention to chronic sorrow in parents of children with CF. However, due to changes in CF diagnosis
and management over the past 20 years, such findings may bear little relevance to parents today.
41 Parenting Children with Cystic Fibrosis
For example, current diagnosis occurs much sooner than parents in these studies, potentially
commencing the cycle of chronic sorrow much sooner than previously reported. Moreover, mothers
were the primary respondents in both studies, representing the research focus at the time.
Therefore, it is possible that findings from mothers are not generalisable to fathers who today, are
possibly much more involved with their children than previously.
Though considered normal responses to diagnosis of CF in an infant, psychological effects of
guilt and chronic sorrow are worthy of close attention because persistent adverse emotions may
have significant effects on both child and parent mental health, and ultimately on a child’s physical
health. Personality, family and other environmental factors influencing feelings of guilt and episodes
of chronic sorrow are important to understand. By identifying mediators and moderators of guilt and
chronic sorrow, improved support services can be offered to parents and their families at
appropriate times, resulting in individualised family support.
2.2.2.3 Parental identity formation and parenting self-efficacy.
There is a dearth of research into parent identity formation, both generally and specifically
in relation to paediatric chronic illness. It has primarily been reported as a psychological construct
associated with adjustment to parenting (Cast, 2004; McBride & Toller, 2011; O'Connor & Barrera,
2014). Parental identity formation, or effects on parental identity, due to a CF diagnosis has not
been specifically addressed; however one study reported parental identity formation in CF in a
qualitative exploration of maternal stressors and coping strategies. Hodgkinson and Lester (2002)
described how parental identity was negatively influenced by the child’s CF diagnosis through a
sense of sacrifice of the traditional parent ideology. This sacrifice took several forms; inability to
return to work and becoming socially isolated; and adjusting to an identity that incorporated a
genetic condition causing a substantial amount of guilt. Coming to terms with being a carrier of
condition inherited by a child was part of accepting a changed identity for the mothers in the study.
The process of identity reformation after CF diagnosis may be similar to O’Connor’s and
Barrera’s (2014) identity disintegration, conceptualised as negative perceptions of parenthood and
the parental role and identity; and identity reintegration, characterised by positive reframing of
parenthood and the parental role and identity in relation to the child’s diagnosis. Parental
characteristics that might indicate whether a parent may experience beneficial or adverse effects on
parent identity are currently not clear because of the lack of clarity about effects of paediatric
chronic conditions on parent identity. Moreover, at what point parents start to shift from identity
reintegration to disintegration or vice versa is ambiguous. This valuable information could be utilised
to determine which parents may be at risk of detrimental outcomes for their parent identity, and on
their subsequent self-esteem and general well-being.
42 Parenting Children with Cystic Fibrosis
Similarly, parenting self-efficacy does not appear to be specifically addressed in the CF
literature, but has been reported as a coping resource/strategy (Gibson, 1998). There has been no
elaboration on this finding since. For example, relevant investigation could include parent and
environmental factors that may be associated with parenting self-efficacy in CF, or what effects
different levels of parenting self-efficacy may have on family functioning or on parental self-esteem.
Psychological conceptualisation of parenting self-efficacy is a personal belief that one will be able to
perform parenting tasks successfully (de Montigny & Lacharité, 2005) and has been shown to be a
critical factor in parenting quality and self-esteem (Coleman & Karraker, 1997; Jones & Prinz, 2005).
Self-esteem has recently been studied in adults with CF (Platten, Newman & Quayle, 2013), but not
with parents. Nor is there investigation into how self-esteem may be tied to parental identity in
paediatric CF, though psychological literature about the relationship between identity and self-
esteem (Cast, 2004; Cowan & Cowan, 1992) does suggest a relationship within the parental domain
of identity.
2.2.3 Parental characteristics moderate child health outcomes.
Improvements in life expectancy have shifted the focus of CF research and clinical care
toward the complex interplay among behavioural, psychosocial, and health outcomes for people
living with CF (Barker & Quittner, 2010). Some parental outcomes have been identified as risk factors
for adverse mental health outcomes in children with CF. For example, reports have demonstrated
associations between maladaptive parenting practices and children’s psychosocial functioning
(Cappelli, McGrath, MacDonald, Katsanis & Lascelles, 1989), and parental depression has been
associated with children’s quality of life (Brucefors, et al., 2015). One of the most robust findings
about parental outcomes affecting child health in CF is that parent and family stress and dysfunction
have an adverse effect on CF outcomes. Patterson, McCubbbin and Warwick (1990) showed that
parental stress, poor parental coping and maladaptive family functioning were associated with poor
health outcomes (body mass index and pulmonary functioning) in children with CF. Further study by
Patterson and colleagues (1993) established results which strongly suggested that a balanced family
coping style, where members tend to their own needs as well as the needs of the child with CF, and
low levels of stress, was positively associated with increased health outcomes (pulmonary
functioning) over 10 years. Similarly, adaptive coping has been shown to influence the physical and
psychological well-being of healthy and CF-affected children (Szyndler, et al., 2005) with positive
effects on growth, nutrition and lung function (Patterson, et al., 1993), and respiratory infections
(Turner-Cobb & Steptoe, 1998).
Parenting practices and parental factors as contributors to child health and development in
paediatric chronic disease are likely to be similar for parents of children with CF. There is also a
potential moderating nature of disease effects on child outcomes. For example, parental stress and
43 Parenting Children with Cystic Fibrosis
depression as a result of CF diagnosis may influence a child’s health outcomes through their effects
on treatment adherence (Barker & Quittner, 2016; Sheehan, Hiscock, Massie, Jaffe & Hay, 2014). A
holistic view of parenting within the family context, inclusive of family functioning and illness
perception as direct and indirect contributing factors to child health are recent assertions that follow
the shift of focus from individual to family-level research and intervention. Given these findings,
there is substantial evidence to support the idea that parenting practices and parental factors
contribute to optimal child health and development in the context of CF. Recent conceptualisations
of how these relationships may develop have been referred to as biological embedding (Hertzman,
1999) and intergenerational or developmental cascading (Masten & Cicchetti, 2010; Patterson,
Forgatch & deGarmo, 2010).
2.2.4 Summary and conclusion.
Increasing life expectancy of children with CF is of paramount importance for clinicians and
biomedical researchers alike. However, the psychosocial environment of the child is also recognised
as an important factor in the health and well-being of the child, both as a moderator and mediator
(Grant, et al., 2006; Robinson, et al., 2008). Therefore, parental psychological well-being is in itself
extremely important, and as such, is the primary focus of my research. This section has detailed how
a CF diagnosis is experienced by parents and how it becomes assimilated into family life. Most
research has focussed on negative aspects of parental experiences of CF. Whilst this research
remains necessary, research that investigates all experiences is essential to understand the full
spectrum of parental experiences of their child’s condition so that negative aspects can be
minimised and positive aspects can be enhanced and taught to others. Similarly, although CF is now
diagnosed early in life, there is insufficient study of the psychosocial effects of CF on children, their
parents and their families in infancy and pre-school. This section has also shown how CF affects
many aspects of behavioural, social and emotional development, which in turn influence health
outcomes and quality of life. Therefore, it is important to consider relationships among multiple
systems (for example, family, peers, health care team), as well as to identify key periods of transition
that require extra monitoring and targeted interventions.
2.3 Coping in families with CF
2.3.1 Coping with diagnosis.
Further to parents’ experiences of their child’s CF diagnosis is the importance of how they
cope. Much empirical investigation regarding parental coping with CF has focused on determining
factors that influence positive and negative coping, that is, adaptive and maladaptive coping
strategies that impede or facilitate adjustment to diagnosis. Research has been conducted across a
number of disciplines, such as nursing, psychology and paediatrics, with influential factors revealed
at societal, group and individual levels. For example, research has found that societal factors such as
44 Parenting Children with Cystic Fibrosis
access to medical services can influence coping. Thomas, Mitchell, O’Rourke and Wainwright (2006),
for example, found that parents in regional areas had less access to CF-specific medical care than
parents living in urban centres, often resulting in feelings of isolation and inadequate support, which
negatively influenced coping strategies.
Maladaptive (or dysfunctional) coping strategies are commonly reported by parents
following CF diagnosis, including strategies of avoidance and denial (e.g., Sheehan, et al., 2014;
Wong & Heriot, 2008). Such maladaptive coping strategies have been associated with low levels of
family supportiveness (Thompson, et al., 1992b), family conflict (Szyndler, et al., 2005), and financial
strain (Goble, 2004). Though maladaptive coping strategies should be addressed by clinicians and
other HCPs, it must be noted that some coping strategies applied on a short-term basis can serve
adaptive purposes while the parents and/or family come to terms with the diagnosis. For example,
cognitive avoidance and distraction may give parents time to adapt to behavioural aspects of
diagnosis and treatment whilst maintaining their psychological well-being (Abbott, 2003). Therefore,
consideration should be given to purposes of particular coping strategies at particular times.
Parental adaptive coping strategies reported by parents during their child’s CF diagnosis
following NBS include problem-solving, seeking social support, searching for information, or not
talking with others about their situation until such time that they had accepted the diagnosis and
could talk comfortably with others about it (Merelle et al, 2003, Tluczek, et al., 2005). These adaptive
coping strategies have been associated with optimism, acceptance, family integration and cohesion,
social support and health literacy (Abbott, Dodd, Gee & Webb, 2001; Harrop, 2007; Hodkova,
Abbott, Mala & Chladova, 2008; Hymovich & Baker, 1985; Liossi & Evans, 2005; McCubbin et al.,
1983; McCubbin, et al., 1984; Pfeffer, et al., 2003; Venters, 1981).
What is evident is that the majority of research into coping with a CF diagnosis has been
conducted with patients and their families prior to the introduction of NBS programs. Whilst some of
the aforementioned research considers children diagnosed via NBS and their families, the number of
these studies is small and research has only recently been conducted. Therefore, it is possible that
psychosocial factors associated with coping identified in older studies prior to NBS no longer bear
relevance to families today. Moreover, most aforementioned studies examined maternal coping
because the majority of participants were mothers where both parents were involved. This may
mean that findings from these studies are not generalisable to fathers who, today, can be central to
a child’s treatment management practice and family intervention.
Finally, there is little recent information to show whether coping styles and strategies of
mothers and fathers of children with CF differ. However, mothers and fathers have been shown to
cope differently with children who have other chronic conditions (Al-Yagon, 2015; Compas, et al.,
2015; Doherty, et al., 2009; Eiser & Havermans, 1992; Heaman, 1995; Wiedebusch, et al., 2010).
45 Parenting Children with Cystic Fibrosis
Specifically for parents of children with CF, an early study reported fathers had lower levels of
adjustment issues such as depression and anxiety than mothers (Thompson & Gustafson, 1996).
However, a recent study reported similar coping strategies between mothers and fathers
(Havermans, et al., 2015). To date, these findings have not been fully explored in fathers of children
who have been diagnosed with CF following NBS. A local study reported fathers’ perspectives of
their child’s CF diagnosis where coping was mentioned by a number of participants within the study.
Fathers commonly reported assuming the protector role within the family, which facilitated their
own, and the mother’s, ability to cope (Priddis, et al., 2010). Therefore, a holistic family perspective
on coping with paediatric CF requires further research into fathers’ coping mechanisms, and
whether they correlate with maternal coping and adjustment.
2.3.2 Coping with CF in the early years.
Research into parental coping with CF once focussed on coping later in a person’s life,
usually adolescence or later (e.g., Gayton, et al., 1977; Gibson, 1988; McCubbin, et al., 1983), and
coping with acute episodes of illness once disease progression had commenced (e.g., Keller &
Nicolls, 1990). Generally, parents of young children with CF no longer need to cope with their child
dying in their early years of life. Instead, they need to cope with a demanding home treatment
regimen. They also have to cope with an early diagnosis and with early knowledge of their child’s
covert disease progression. Therefore, generally marked by a period of parent and family stress, an
initial period of change and adaptation, both psychologically and behaviourally, is required (Glasscoe
& Smith, 2008; Quittner, et al., 1992). During this phase of stabilisation, parents must learn new
caregiving tasks and treatment regimens (Quittner, et al., 2009). It is during this period when
families will build foundations for how they will cope with the condition during childhood and
adolescence (Rolland, 1987), which are important for both current and future management of the
disease. Following research trends into parenting children with chronic conditions outlined in the
introductory chapter, research into parental coping in the context of paediatric CF initially focussed
on psychopathology with the aim of intervening and reducing maladjustment. Venters (1981) was
one of the first to report on parental coping with a child’s CF. She identified coping strategies that
minimised condition-related hardships were statistically significantly associated with long-term
adequacy of family functioning.
Over the past 30 years, there has been an abundance of research published on parental and
familial coping in CF (Abbott, et al., 2001; Abbott & Gee, 2003; Cappelli, et al., 1988; Cowen, et al.,
1986; Coyne, 1997; Hodgkinson & Lester, 2002; Hovey, 2005; Lewis & Khaw, 1982; McCubbin, et al.,
1984; Nagy & Ungerer, 1990; Patterson, et al., 1993; Pendleton, Cavalli, Pargament, & Nasr, 2002;
Quittner, et al., 1992; Quittner, et al., 1996; Spirito, Stark, Gil, & Tyc, 1995; Staab et al., 1998;
Thompson, et al., 1992b; Wong & Heriot, 2008). From these studies, it is has been shown that
46 Parenting Children with Cystic Fibrosis
families generally experience a period of uncertainty and disruption after diagnosis before returning
to relatively common and conventional levels of family functioning and adaptability.
Many parental coping strategies have been reported in the literature. Some of the more
dominant strategies include maintaining optimism (Abbott, 2009; Abbott, et al., 2001; Bauchner, et
al., 1996; Hodgkinson & Lester, 2002; McCubbin, et al., 1984), cognitive restructuring (Hodgkinson &
Lester, 2002; Thompson, et al., 1992b), maintaining and/or seeking social and emotional support
(Hodgkinson & Lester, 2002; Hymovich & Baker, 1985; McCubbin, et al., 1984; Thompson, et al.,
1992b; Wong & Heriot, 2008), seeking medical information from other parents or HCPs (Hovey,
2005; Hymovich & Baker, 1985; McCubbin, et al., 1984; Thompson, et al., 1992b), cognitive
avoidance (Abbott, et al., 2001; Sheehan, et al., 2014; Staab, et al., 1998; Wong & Heriot, 2008) and
distraction (Abbott, 2009; Abbott, et al., 2001).
A recent Australian study reported moderate correlations of parental depression, anxiety
and stress with avoidant coping (Sheehan, et al., 2014). Avoidant coping has three aspects; focusing
on venting of emotions, behavioral disengagement, and mental disengagement (Carver, Scheier &
Weintraub, 1989). Avoidant coping strategies generally have little or no effect (Roth & Cohen, 1986).
Furthermore, avoidant coping was significantly correlated with eating and mealtime problem
behaviours in children (Sheehan, et al., 2014). What this has the potential to explain is that parental
avoidant coping behaviours can contribute to children’s dietary complications, which can result in
low nutritional status. Therefore, it appears that parental avoidant coping can influence low
nutritional status, which might be mediated by mealtime and eating complications. Collectively,
studies investigating parental coping strategies identify both problem and emotion-focussed coping
styles and strategies (explained in the next chapter), with emotion-focussed strategies being less
effective, which is analogous to the general coping literature (e.g., Li, Cooper, Bradley, Shulman &
Livingston, 2012; Regier & Parmelee, 2015; Shin, et al., 2014).
Similar to investigation of parental coping with a CF diagnosis, the majority of research into
parental coping with CF in the early years of a child’s life has focused on children and their families
prior to the introduction of NBS programs. Therefore, it is possible that coping strategies and their
effectiveness identified in studies prior to NBS are no longer appropriate reports of parenting
children with CF. Moreover, most studies failed to assess the effect of development and lifespan
issues on parental experiences, collapsing across child age, generally ranging from 1 to 16 years.
Categories of age ranges for the youngest group generally consisted of children from birth to 6 years
of age, which could result in unintentional disregard for developmental issues that may impinge on
parental coping resources and other important experiences.
One coping strategy distinct to fathers of children with CF has been reported. Fathers appear
to have lower self-competency about caring for their child than mothers and this was magnified by
47 Parenting Children with Cystic Fibrosis
the mother as ‘expert.’ This feeling of low self-competency was counteracted by mastery of the
treatment regimen, which allowed fathers to be involved in the caring process and therefore feel a
sense of adequacy (Goldberg, et al., 1990). So although most research is on maternal coping, fathers
have been shown to cope differently, which mirrors literature in other chronic diseases (e.g., Barak-
Levy & Atzaba-Poria, 2013; Compas, et al., 2015; Twoy, Connolly & Novak, 2007). This is of great
value to researchers, clinicians and the broader medical and psychological communities because if
necessary, tailored intervention strategies for improved parental outcomes may be implemented.
2.3.2.1 Resilience and hope in the face of adversity.
In the past 10 years, research has trended towards a focus on resilience factors as mediators
of adjustment and adaptation to CF (e.g., Grossoehme, Szczesniak, Dodd & Opipari-Arrigan, 2014;
Mc Hugh, Mc Feeters, Boyda & O’Neill, 2016); Mitsmansgruber, et al., 2015). As such, current coping
research investigates factors within a parent’s capabilities that reinforce resilience, with the aim of
increasing personal and family strength against the illness stressor as well as associated and future
stressors. Therefore the thrust of knowledge about resilience in parents of children with CF has been
conducted under the auspices of coping research. Resilience is a supplementary term related to
coping (Fletcher & Sarkar, 2013). Whilst they are overlapping terms in the psychological trauma and
loss literature, resilience generally refers to “a dynamic process encompassing positive adaptation
within context of significant adversity” (Luther, Cicchetti, & Becker, 2000, pp. 543). Two critical
components of resiliency are; exposure to significant threat, and achievement of positive adaptation
despite major assaults on the developmental process (Luthar & Zigler, 1991; Masten, Best &
Garmezy, 1990). Due to its modern interest by researchers compared to the coping construct, much
less literature exists about the resilience construct.
Before discussing current literature on resilience as a psychological construct, hope despite
facing adversity is a psychological construct identified as a parental coping strategy contributing to
development of resilience for future burden of physical and mental chronic conditions (Barlow &
Ellard, 2006; Faso, et al., 2013; Horton & Wallander, 2001; Kashdan, et al., 2002; Lloyd & Hastings,
2009; Ogston, Mackintosh & Myers, 2011; Petersen & Wilkinson, 2015; Samson, et al., 2009). Hope
is a motivational factor helping to initiate and sustain action towards goals despite obstacles that
might get in the way of goal attainment (Snyder, Rand, & Sigmon, 2002). In the context of parenting
a child with a chronic condition, hope has been referred to as a transitional refocusing from a
difficult present to a positive future with dynamic possibilities within continual uncertainty
(Duggleby, et al., 2010). Therefore, hope exhibits distinct features in intentionality and agency. That
is, being hopeful refers to an ability to conceptualise goals, find pathways to these goals despite
obstacles and having the motivation to use those pathways (Snyder, et al., 2002). For these reasons,
48 Parenting Children with Cystic Fibrosis
hope has been associated with not only adaptive coping strategies for preventing and confronting
stress, but also a sense of control over achieving one’s goals (e.g., Snyder, et al., 1991).
Little research has been conducted on the lived experience of hope among parents of
children with a chronic, incurable and potentially fatal condition (Samson, et al., 2009). However,
Hodgkinson and Lester (2002), and Gjengedal and colleagues (2003), along with Jessup and
Parkinson (2010) have reported hope as a prevalent emotion-focussed coping strategy for parents of
children with CF. In these studies, hope manifested in both short and long-term form; short-term
hope was for immediate good health for their child and long-term hope was for a cure to the
condition. Hope has been shown to predict reduced anxiety and less emotional impact for parents of
children with CF (Wong & Heriot, 2008). Hope does not necessarily come so easily for parents; at
times parents felt they had to fight to experience hope (Jessup & Parkinson, 2010) or that they were
going through the motions because there was no other alternative but to hope (Hodgkinson and
Lester, 2002). An implication of these findings is that reduction in anxiety and emotional impact of
illness resulting from hope is supportive of developing resilience.
Despite enormous treatment burden and potentially shortened mortality, parents of
children with CF are generally resilient, with most reporting a high quality of life and normative
levels of psychopathology (Brucefors, et al., 2015; Quittner, et al., 2014; Szyndler, et al., 2005).
Resiliency research has primarily emanated from the loss and trauma literature, predominantly
focusing on what makes people resilient in the face of personal, economic and environmental
adversity (see discussions in Agaibi & Wilson, 2005, and Richardson, 2002). A resiliency approach to
understanding outcomes of adversity entails exploring potential for personal and relational growth
that can be generated from adversity (Boss, 2001). By tapping into key processes for resilience,
individuals and families can emerge stronger and more resourceful to meet future challenges
(Walsh, 2003). This approach to understanding the family system and its individual members is
consistent with the salutogenic orientation first proposed by Antonovsky in 1987 and more recently
by Seligman and Csikszentmihalyi (2000) in the positive psychology movement.
As a result of the transition from a psychopathologic view on paediatric chronic illness,
resilience has become an important concept in mental health theory and research over the past two
decades (Rolland & Walsh, 2006). Current foci of resilience in context of paediatric chronic
conditions is the complex interplay of multiple risk and resistance factors including individual, family
and broader socio-cultural influences. From a family perspective, concentrating on strength in the
face of adversity has shown that the role of family is nurturance and development, and its function is
management and cohesion of individual family members within a synergistic and dynamic system
(Patterson, 2002; Walsh, 2003). Within the family system, protective factors have been identified
49 Parenting Children with Cystic Fibrosis
that build and foster resilience including support and communication, family meaning-making, locus
of control and self-efficacy, and effective coping skills (Black & Lobo, 2008).
Support is a resiliency factor for parents managing their child’s paediatric condition (Benzies
& Mychasiuk, 2009; Peer & Hillman, 2014). Instrumental, social and practical forms of support assist
families during times of stress and help them to maintain optimal mental and physical health
(O’Leary, 1998; Walsh, 2003; Black & Ford-Gilboe, 2004). Therefore, formal and informal supports
buffer parents against negative effects of stress associated with their child’s condition (Peer &
Hillman, 2014). Communication as a source of support also helps family members to work through
and understand emotions and cognitions associated with stressful events (Armstrong, Birnie-
Lefcovitch & Ungar, 2005; Finkenauer, Engles, Branje, and Meeus, 2004; Kelly & Emery, 2003).
Communication within, and outside of, the family can also help families to make meaning of adverse
events.
Additionally, it is well-known that how one appraises an event is a determining factor of how
one will cope with the event (Lazarus & Folkman, 1984). Based on this premise, belief systems are
vital for family resilience because how a family appraises, and makes meaning of, an event will help
determine how they cope. Making meaning of an adverse event can change perspective of how the
adversity is viewed. For example, families can develop a sense of coherence (i.e, motivation for the
family to stick together) by recasting a crisis as a challenge that is manageable and meaningful to
tackle (Antonovsky & Sourani, 1988). Sense of coherence refers to the extent to which families feel
confident that the outcomes of a situation will be favourable for them (Hawley, 2000). Similarly,
generating a positive outlook in the face of adversity functions to conserve a family’s energy and
efforts in managing the adversity (McCreary & Dancy, 2004; Orthner, Jones-Sanpei, & Williamson,
2004; Peer & Hillman, 2014; White, Richter, Koeckeritz, Munch & Walter, 2004). Individual factors
have also been found to have an important protective function that contributes to family resiliency.
Individual protective factors that contribute to family resilience include locus of control,
which refers to an individual’s perceptions of where underlying responsibilities for life events resides
(Rotter, 1966). A dichotomous system of internal and external loci of control exists to explain where
responsibility lies. An internal locus of control orientation is a belief about whether the outcomes of
actions are contingent on what a person does, whereas an external locus of control orientation is a
belief that events are outside of personal control (Zimbardo, 1985). Those with an internal locus of
control exhibit empowerment and agency, and are willing to put in the effort required to make
positive changes in the face of adversity (Juby and Rycraft, 2004).
Related to locus of control is self-efficacy, which refers to an individual’s judgment of their
own ability to succeed in reaching a specific goal (Bandura, 1994), for example, parenting self-
efficacy. Being able to act and think independently and confidently are important individual
50 Parenting Children with Cystic Fibrosis
protective factors (Place, Reynolds, Cousins & O’Neill, 2002). Self-efficacy has been reported as a
strong predictor of family health promotion (Black & Ford-Gilboe, 2004). Lastly, effective coping
skills of individual family members have a direct impact on the family resiliency process (Benzies &
Mychasiuk, 2009). Coping is a response to demands appraised by a person as taxing or exceeding
available resources (McCubbin & McCubbin, 1993). For example, effective coping skills have
reportedly buffered against financial strain (Lloyd & Rosman, 2005) and mediated the relationship
between maternal depression and child behavioural problems (Lee, 2003).
Common pathways to family resilience, such as communication through support and making
meaning through appraisal, are important to understand and harness. Similarly, characterising
individual protective factors important for family resilience offers a starting point for development
of clinical intervention. Therefore, developing a pathway model of individual and family
characteristics and processes that lead to resilience would allow characterisation of how families
might respond to adversity, as well as development of targeted interventions that can be tailored to
family needs. Collectively, the above-mentioned findings demonstrate clinical utility of focussing on
family resilience in the context of paediatric chronic illness by; building individual and family
strength, searching for commonalities among diverse pathways to resilience, and emphasising
usefulness of developing effective coping strategies and positive family-level schemata.
2.3.3 Summary and conclusion.
The available psychosocial research into CF has shown how parents cope with their infant’s
diagnosis and how families cope with CF and associated family experiences. Various studies report
that after initial disruption and adjustment, families continue to function well when a child is
diagnosed with CF. These findings contrast with reports of consistent, cyclical negative effects on
parents of children with CF such as anxiety, stress, depression and PTSS symptoms, particularly for
mothers. Therefore, screening for potential psychosocial issues can assist clinicians to identify and
understand barriers that parents and families encounter. Education and enhancing beneficial
psychosocial experiences and outcomes for parents can assist them to manage CF within their family
and to cope with their child’s early surveillance of CF lung disease.
2.4 Aims and significance of this research
2.4.1 What does this study hope to achieve and why is it important?
With the implementation of NBS for CF in Australia, surveillance of disease progression and
prophylactic treatment for children and infants with CF has been possible. This has led to the
introduction of AREST CF, which conducts research in the detection, prevention and treatment of
early lung disease in CF. AREST CF has now been operating in Australia for over 10 years. Whilst
there is now some research into the psychological effects of a CF diagnosis via NBS, there is a dearth
of research into psychological effects of early surveillance and potential consecutive prophylactic
51 Parenting Children with Cystic Fibrosis
treatment following a CF diagnosis via NBS, and no research assessing parental psychological effects
of the AREST CF program. Although NBS for CF is arguably beneficial, discussion and debate needs to
continue to maximise beneficial outcomes for children, their parents and their families. This should
include clinicinas and other HCPs coming together in subsequent treatment programs following a
positive diagnosis of CF via NBS to maximise beneficial outcomes for family members who have a
child diagnosed with CF.
As little is known about parents whose children undergo early surveillance for CF lung
disease, the purpose of my research is to explore the lived experience of parenting in the context of
early surveillance for CF lung disease and to describe how parents cope with their experiences.
Moreover, using a strength-based approach to understanding parenting practices and parental
characteristics in the current context will identify protective factors that can be enhanced for those
possessing them and educated to others who don’t. Lastly, exploring how early surveillance affects
parental constructions of CF can distinguish unique parental outcomes that result from such a
program, informing institutions of how such a program is interpreted by parents. Exploring how
parents construct their experiences and attribute meaning to early surveillance, and to their child’s
condition in the context of early surveillance, will lay a foundation from which necessary clinical
intervention can be developed that comprises a family perspective. Therefore this research may
guide future clinical interventions aimed at improving mental health and well-being for parents
managing their child’s chronic condition in the context of early surveillance and facilitate further
holistic research that includes all family members who are affected by a child’s chronic condition.
2.4.2 How can this research help families whose children undergo early CF disease
surveillance?
Although there may be some parallels between day to day experiences and coping with CF
and with early surveillance, several clinical components of early surveillance may elicit different
parental responses and require additional coping strategies and skills that have yet to be
understood. Insights from related fields also indicate that anxiety, stress, and depression are the
most commonly reported outcomes for parents with a chronically ill child. Therefore, it is important
to investigate whether these negative experiences and mental health morbidities occur for parents
of children engaged in early surveillance. By identifying if parents in the current context experience
additional negative outcomes as a result of their child’s early surveillance, it is imperative that AREST
CF ensures parents are supported. As AREST CF is a unique research and clinical program, we must
ensure that unique experiences and outcomes are explored and if need be, intervened. Of equal
importance is ensuring that beneficial outcomes are understood so that parents’ lives can be
optimally enhanced. My research will have a specific focus on coping and adaptation as these are
dominant topics of research in CF with plenty of published studies, but none examining coping with
52 Parenting Children with Cystic Fibrosis
CF in the context of early surveillance. Improved psychosocial outcomes for CF patients and their
families can lead to improved physiological outcomes for the patient, and long-term beneficial
outcomes for the entire family.
Therefore, whilst early surveillance informs parents and HCPs of the covert changes in lungs
that are not detectable through conventional methods, understanding what parents experience,
how they cope, and exploring psychosocial factors associated with their experiences and coping
styles is essential to inform CF centres and researchers considering adopting this program. There is
also a need to inform intervention strategies that aim to support the family and foster healthy
coping mechanisms. As it is not yet known which psychosocial factors in a child’s environment
influence coping and other parental experiences during early surveillance, the findings should assist
with identifying risk and protective factors for negative psychosocial outcomes and resiliency.
Therefore, my research outcomes will guide clinical screening for known psychosocial and familial
risk factors, and facilitate building family resiliency for future burden of CF, hence guiding the design
of individualistic support for families.
2.4.3 What else can this research add to the body of knowledge?
My research contributes to a growing body of knowledge about parenting children with
chronic conditions. Uniquely, findings from this research offer insight into the lived experience of
parenting children with CF in the context of early surveillance; with this information assisting in
understanding holistic needs of parents, particularly those in Western Australia and Victoria. In
practical terms, identification of psychological factors important in maintaining optimal functioning
and quality of life, and insight into constructions of experience and attributions of meaning given to
CF in the context of early surveillance, may be used to inform existing and future policy decision-
making models for best clinical practice towards promoting mental health and well-being.
Accordingly, this research informs existing and future policy structures through identification of
previously omitted information or misconceived assumptions regarding parenting a child undergoing
early surveillance and the issues facing them. Moreover, my research contributes to understandings
of parenting children with chronic conditions as a process of normalisation, management, resilience
and reconstructing future optimistically through hope rather than focussed solely on deficits
typically associated with parenting children with chronic conditions. Foremost, the findings
contribute to building a holistic understanding of the parenting experience and provide evidence
that parenting a child with CF in the context of early surveillance for lung disease has both adverse
and beneficial outcomes that are amenable to modification. Consequently, this research; is entirely
original, is based on sound theoretical and empirical concepts, contributes new knowledge relevant
to the global CF community, and provides a foundation for new clinical pathways that support
53 Parenting Children with Cystic Fibrosis
patients and families. The next chapter presents theoretical underpinnings and methodological
decisions for conducting the research.
54 Parenting Children with Cystic Fibrosis
CHAPTER 3
Substantiative Theoretical and Methodological Derivatives
This chapter outlines the rationale for theoretical and methodological choices underlying the
process and implementation of the research. Firstly, theoretical models chosen through
which to view the findings are reviewed in reference to how they have informed the
research by outlining family resiliency, adaptation, and stress and coping approaches.
Secondly, chosen theoretical models are critiqued as replacement of the once dominant
psychopathological view to parenting a child with CF. This leads to an introduction of the
research design and the qualitative methodology used in this study. Specifically,
phenomenological inquiry is reviewed in relation to how it informed the research.
Methodological justifications are further described for data collection and sampling
procedures. The chapter concludes by substantiating the thematic data analytic technique.
55 Parenting Children with Cystic Fibrosis
He who loves practice without theory is like the sailor who boards ship without a rudder
and compass and never knows where he may cast...Leonardo da Vinci
3.1 Introduction
Viewing research findings through theoretical lenses allows a broad application of the
findings beyond their current context, as well as providing an opportunity to support or refute a
model’s components within the findings’ current context. Accordingly, this chapter initially outlines
the rationale for chosen theoretical models through which to view my research findings. Following
that is a description of the grounds for methodological decisions made to implement the study.
Presenting substantiative evidence of the theoretical and methodological choices allows the reader
to determine authenticity and appropriateness of how the study was conducted prior to learning
about the operational aspects. The theoretical decisions are primarily based on literature presented
in the previous chapter, whereas methodological decisions were based on the nature of the study.
Given the observation that the majority of families faced with care of a child with CF do not
appear dysfunctional and are, in fact, coping successfully with a psychologically and logistically
challenging situation, a contextual stress and coping conceptual framework has been developed
(McCubbin & McCubbin, 1987; 1993). The framework was initially developed with families of
children with CF, and provides a set of lenses through which to view these families. The shift in focus
purely from parental psychopathology to beneficial psychological outcomes is exemplified by a
model commonly cited to explain relationships among adaptive behaviours, family support,
parenting stress and coping in the context of chronic illness. The Resiliency Model of Stress, Family
Adjustment and Adaptation (McCubbin & McCubbin, 1987; 1993) arose from the notion that families
not only react to stress, but they attempt to navigate their way through stresses to achieve
adjustment, and eventually adaptation. Theoretical underpinnings of the Resiliency Model are
grounded in Lazarus’ and Folkman’s (1984) Transactional Model of Stress and Coping whereby three
variables are present in a situation that determines whether a crisis is created or not; the hardship of
the situation; resources required to meet the situation; and appraisal of the event as either neutral
or threatening. Lazurus’ and Folkman’s theory is a theoretical structure through which to explore
and investigate cognitive appraisal of a stressful event and activation of coping resources as a
response, which lays the foundation for how the Resiliency Model accounts for parental outcomes
as a result of managing a chronic condition within the family environment.
3.2 Theoretical frameworks
3.2.1 Transactional Model of Stress and Coping.
Lazarus’ and Folkman’s Transactional Model of Stress and Coping (1984) has led to
understanding and explaining stress and coping within the health sciences. Their cognitive model has
defined coping as thoughts and behaviours people use to manage internal and external demands of
56 Parenting Children with Cystic Fibrosis
situations that are appraised as stressful. The coping process is initiated in response to an
individual’s appraisal that important goals have been harmed, threatened or lost. These appraisals
are initially characterised by negative emotions and are therefore initiated in an emotional
environment. One of the first coping tasks is therefore, to down-regulate (i.e., reduce) negative
emotions that are stressful and may interfere with instrumental (i.e., behavioural) forms of coping.
Emotions continue to be fundamental to the coping process throughout a stressful event or situation
as an outcome of coping, as a response to new information, and as a result of reappraisals of the
status of the stressful event or situation. If the stressful event or situation has a successful
resolution, positive emotions will ensue; if the resolution is unfavourable or unclear, negative
emotions will likely ensue (Folkman & Moskowitz, 2004).
According to Lazarus (1966), the individual difference of most relevance to the study of
human behaviour and stress is the cognitive appraisal of stressors. His early theory placed attention
on the role of cognitive appraisal in producing the quality of the individual’s emotional response to a
stressful encounter or event, and the ways in which a person coped with the appraised encounter or
event. Lazarus believed that after appraising the stressor, an individual will use one or more coping
strategies in an attempt to adjust to the situation. Similarly, which coping responses emerge is partly
determined by an individual’s knowledge of coping options and partly by their beliefs about the
usefulness of those options. Both stress and coping then, emanate partly from the mental sets
brought by the person to the event (Parker & Endler, 1996). Cognitive appraisal then became a
critical aspect of stress research methodology and theory that respected individual differences in the
nature and degree of stressful experiences (Crnic & Low, 2002). Cognitive appraisals refer to direct
and immediate evaluations about the individual’s environment with reference to their well-being.
They act as evaluative frameworks that individuals utilise to make sense of events (Lazarus &
Folkman, 1984). Emphasis on cognitive appraisal as a contributor to psychological outcomes has
continued into the context of parenting stress (Deater-Deckard, 1998).
Two main coping dimensions studied in early literature about coping, and that have since
received much research attention, were emotion-focussed coping and problem-focussed coping. In
summary of the sizable literature in this area, problem-focussed coping involves strategies that
attempt to solve, reconceptualise, or minimise the effects of stressful events. On the other hand,
emotion-focussed coping includes strategies that involve self-preoccupation, fantasy, or other
conscious activities related to emotion regulation (Parker & Endler, 1996). Avoidance-oriented
coping has also been identified as another basic dimension that has received much research
attention, and has been conceptualised as involving person-oriented and/or task-oriented
responses. An individual can react to a stressful event by seeking out other people (i.e., social
diversion) or can engage in a substitute task (i.e., distraction).
57 Parenting Children with Cystic Fibrosis
Research into coping has demonstrated some consistent findings. Well-established findings
include both dispositional and situational attributes heavily influence coping behaviours (Bouchard,
Guillemette & Landry-Léger, 2004; Carver & Scheier, 1994; Panayiotou, Kokkinos & Kapsou, 2014).
Coping is strongly associated with the regulation of emotion (especially distress) (Webb, Miles &
Sheeran, 2012). Some coping strategies (e.g, avoidance coping strategies) are consistently associated
with poor mental health outcomes (Dardas & Ahmad, 2015; Meier, Carr, Currier & Neimeyer, 2013).
Collectively, these findings suggest that individuals who are flexible in their choice of coping
strategies should show better adaptation than individuals who have a more restricted coping
repertoire.
With convergent evidence about the applicability of coping research being produced,
research has considered beneficial outcomes of stress and coping. Some research has considered
positive outcomes of successfully resolving a stressful encounter (e.g., Folkman & Lazarus, 1985),
including outcomes such as acquisition of new coping skills and resources (Schaefer & Coleman,
1992), and the perception of growth related to the stress (Holahan & Moos, 1990). Other research
has considered the frequency of positive affect during stress (e.g., Viney, 1986; Folkman, 1997), and
found that positive emotion does in fact co-occur with negative emotion related to stress.
Since early conceptualisations of coping, a proliferation of research has developed across
behavioural and social sciences, medicine, public health and nursing (Folkman & Moskowitz, 2004).
Current understandings of coping include a multi-dimensional process that is sensitive both to the
environment, and its demands and resources, and to personality dispositions that influence the
appraisal of stress and resources for coping (Somerfield & McCrae, 2000). Coping research in
specialised areas, such as parenting, is the result of an attempt to try and combat some of the issues
raised in general coping research and to make further generalisations about a particular population,
a particular stressful encounter or event, or a particular population encountering a particular
stressful situation or event.
3.2.2 The Resiliency Model of Family Stress, Adjustment and Adaptation.
The shift in focus from psychopathology to adaptive psychological outcomes of a paediatric
chronic condition, particularly from a family perspective, resulted in a conceptual model to
understanding relationships among adaptive behaviours, family support, parenting stress and
coping. Moreover, if one accepts the basic observation that most families faced with care of a child
with CF do not appear dysfunctional and are, indeed, coping successfully with a difficult situation, a
resiliency framework can provide another set of lenses through which to view these families. The
Family Resiliency Model of Stress, Adjustment and Adaptation (McCubbin & McCubbin, 1993) has
had a number of iterations, beginning with a strong focus on psychopathology within the ABC-X
model of family behaviour (Hill, 1949; 1958) and later, the Double ABC-X model of family behaviour
58 Parenting Children with Cystic Fibrosis
(McCubbin & Patterson, 1983). In the early model, A referred to the crisis-precipitating event, B
referred to the family’s crisis-meeting resources and C referred to the definition the family makes of
the event via their appraisal mechanism (Hill, 1958). A shift in emphasis from crisis to transition
resulted in the Resiliency Model’s emphasis on adaptation as the most often needed response to a
chronic condition within a family context.
The current model identifies essential interacting components of family life and explains, as
well as predicts, family processes and outcomes of managing a paediatric chronic condition. The
model outlines how these outcomes can vary along a continuum of extremely positive
(bonadjustment and bonadaptation) to the more negative extreme of maladjustment and
maladaptation. The length of time one has a chronic condition means that one’s family must
attempt to adjust immediately, and to adapt to the condition on a more permanent basis over time.
Therefore, the model consists of two phases. The first phase is an adjustment phase, characterised
by relatively minor changes in the family system, and is a predictable phase in the family’s response
to a stressor such as an illness. The second phase is an adaptation phase, a slower, more disruptive
and resistant type of adjustment. Each phase has inter-related components combining to produce
positive or negative outcomes. The model attempts to explain the processes families must negotiate
and the objectives that must be accomplished to achieve bonadaptation. Bonadaptation refers to a
family’s ability to meet the needs of each individual family member, enabling them to achieve their
maximum potential as well as the functioning of the family system and its transactions with the
community (McCubbin & McCUbbin, 1993). Accordingly, the model serves as a framework for
determining whether or not a health or illness stressor becomes a crisis. See the following page for a
diagram of the constructs, processes and relationships that comprise The Resiliency Model of Family
Stress, Adjustment and Adaptation.
59 Parenting Children with Cystic Fibrosis
Figure 1. The Resiliency Model of Family Stress, Adjustment and Adaptation. Adapted from “Families, Health and Illness: Perspective on Coping and Intervention,” by H. I. McCubbin and M. A. McCubbin, 1993, p. 23. Copyright 1993 by Mosby.
60 Parenting Children with Cystic Fibrosis
The Resiliency Model (McCubbin & McCubbin, 1993) has extracted theoretical
underpinnings from a number of other models attempting to explain similar phenomena from a
more generalised perspective, that is, within non-context specific events or circumstances. Firstly, a
resiliency approach to family health is consistent with the broad biopsychosocial model (Engel, 1977)
in that it theorises that health is the result of multiple and interacting influences, including risk in the
environment and protective processes available to the family. Secondly, the impact of illness on
family coping skills is explored (including successful and unsuccessful coping characteristics). It
highlights the reciprocal relationship between physical and mental health and family functioning,
thereby supporting Family Systems theory (Bowen, 1978) underpinning the model for understanding
stress and the impact of illness on the family. Lastly, Lazarus’ & Folkman’s (1984) Stress and Coping
theory’s concept of appraisal underpins the evaluation (i.e., appraisal) of, and subsequent response
to, the family stressor of paediatric chronic illness demonstrated in the Resiliency Model’s individual
level and family schema level of appraisal. The Resiliency Model is therefore a comprehensive and
context-specific theoretical model relevant for guided investigation of familial coping and adaptation
to paediatric chronic illness - and specifically for the purpose of my thesis, isolating parental coping
and adaptation to paediatric CF.
Notwithstanding the model’s strengths in exploring parenting and family functioning in the
context of paediatric chronic illness, theoretical models nonetheless possess areas for improvement
or empirical support for their justification. The Resiliency Model (McCubbin & McCubbin, 1993)
posits that the ultimate key to determining the direction of a family’s response to an illness stressor
is the family’s situational appraisal that governs, if any, of the other illness dimensions are
problematic to a family. Individual-level appraisal is well-documented, however empirical
investigations of family-level appraisal seem to be limited, and could therefore benefit from further
investigation into the applicability of this dimension. Family-level analysis is invaluable to understand
and support family functioning but it fails to identify any nuances between family members and
within individual family members that function either synergistically or antagonistically within the
family. Therefore, whilst the model is adept at identifying family-level factors important for
developing and maintaining resilient families, individual and dyadic factors associated with resilience
should be of primary consideration before embarking on a family-level analysis. Moreover, whilst
the Resiliency Model was developed with data collected from families with a young child with CF,
the model is primarily theoretical in its conceptualisations. Research has supported its theoretical
use (DeHaan, Hawley & Deal, 2013; Hawley, 2000; Simon, Murphy & Smith, 2005), and research
demonstrating its clinical application is still in its infancy (Frain, et al., 2007; Gardner, Huber, Steiner,
Vazquez & Savage, 2008; Paynter, Riley, Beamish, Davies & Milford, 2013; Tak & McCubbin, 2002).
61 Parenting Children with Cystic Fibrosis
Within the scope of my thesis, concepts within the Resiliency Model (McCubbin &
McCubbin, 1993) may begin to indicate how parental processes can influence, and be influenced by,
systemic, family and personal factors. Parental processes affect parental coping of, and ultimately
adaptation to, CF specifically, and paediatric chronic illness more generally. Whilst, according to the
model, there are many factors that contribute to family adaptation (as seen in Figure 1), processes
and outcomes specific to parents’ ability to cope with (i.e., adapt to) their child’s chronic condition
will be the topic of the remainder of the current discussion.
A well-known study by Patterson (2002) investigated families of children with CF and
emphasised two key features of family life that serve as protective factors under challenging
conditions, both of which are inherent in the Resiliency Model. The first key feature is family-
meaning making, or the family’s creation of their world-view. Families vary considerably in their
interpretations of health and illness, and develop strategies according to their beliefs. Therefore, the
family belief system can influence how it manages daily demands and response to the accumulation
of stressors over time. That is, the appraisal or meaning of an event can promote healthy adaptation
to stressful conditions. The influence of Lazarus’ and Folkman’s (1984) work underpins this process.
Confirmation of appraisal and meaning-making as contributing factors to how a family adapts to CF
is work done by Baldacchino, Borg, Muscat & Sturgeon (2012) who showed that appraisal of, and
meaning attributed to, a child’s CF influenced the way parents chose to cope with it. They identified
that the more positive the parents’ appraisal of their child’s condition, the more adaptive their
coping mechanisms.
The other key feature of the Resiliency Model (McCubbin & McCubbin, 1993) involves
establishing newly instituted patterns of family functioning. Adaptive patterns of family functioning
promote healthy behaviours by providing an organisational structure and predictability to family life,
and provide an element of planning ahead and role assignment. The outcomes of adaptive family
functioning include family cohesion, understanding and acceptance of individual family roles, and
development of self-efficacy in maintaining adaptive functioning (Ciao, Accurso, Fitzsimmons-Craft,
Lock & Le Grange, 2015; Crerand, et al., 2015; Tramonti, et al., 2015).
One such adaptive pattern attributed to effective parental coping and increasing family
stability is normalisation of the chronic condition into family life. Normalisation of the disease within
the family is a prevalent coping mechanism for families of children with CF (Gibson, 1988, Glasscoe &
Smith, 2011; Hodgkinson & Lester, 2002). Normalisation has also been expressed as new normal
whereby some patients created a new sense of normality, balancing life before and after being
diagnosed (Liu, Inkpen, & Pratt, 2015).
Research into parental coping with the early CF management has reported adaptive
strategies such as establishing a routine that incorporates treatment into daily life, allowing the
62 Parenting Children with Cystic Fibrosis
family to achieve a sense of normality (Bluebond-Langner, 1996; Glasccoe & Smith, 2011;
Grosssoehme, et al., 2014; Hodgkinson & Lester, 2002; Jessup & Parkinson, 2010; Pfeffer, et al.,
2003). Applying the Resiliency Model (McCubbin & McCubbin, 1993) is a purposeful approach to
understand how families establish CF management routines, and to identify mechanisms and
outcomes of family routines. The Resiliency Model has established links between routines and other
family processes, which can be used as a framework to understand outcomes of family routines.
Though establishing family routines have been associated with improved adherence and
health outcomes in other disease populations (e.g., Fiese & Wamboldt, 2003), routines in families of
very young children with CF have not been comprehensively examined. A recent study sought to
explore parental experiences of families with young children in developing and utilising CF care
routines within the family setting (Grossoehme, Filigno & Bishop, 2014). Phenomenological
methodology allowed themes to emerge describing how families of children with CF aged 3 months
to 13 years utilised routines to manage CF care. How families used support structures to facilitate
maintenance of routines, and how families encountered, and overcame, challenges with maintaining
care routines also emerged.
Along with family resources, and based on meaning-making through appraisal and newly
instituted patterns of family functioning, how families problem solve and cope will then determine
their level of adaptation to their child’s chronic condition. Unfortunately, many studies of children
with CF and their families include participants across large, undifferentiated age ranges of children.
Whilst this increases generalisability of the study, it undermines both child and family
developmental processes and achievements. Therefore, it would be interesting to know at what
point of the child’s life that families generally establish such routines so that developmentally-
appropriate guidance can be offered to families that facilitate use of routines and improve CF
management.
Despite the abundance of literature into parental coping with CF in the 1980s, less research
has recently evolved in the literature. Movement from traditional psychopathological views of how
CF is generally assimilated into the family to approaching family adaptation to CF from a resilience
perspective has been well-received. However, technological and medical advancements in CF
continue at a rapid pace, which may mean that techniques and interventions for effective CF
management (such as those offered by AREST CF) and psychological well-being of the patient and
their family may not bear relevance today.
3.2.3 Summary and conclusion.
Theoretical models provide a structural guide for exploring and identifying variables of
interest and their potential associated relationships and outcomes. Commonly-cited theoretical
models applicable to the empirical literature presented in the preceding chapter are The
63 Parenting Children with Cystic Fibrosis
Transactional Model of Stress and Coping (Lazarus & Folkman, 1984) and The Resiliency Model of
Stress, Adjustment and Adaptation (McCubbin & McCubbin, 1993).
The Transactional Model of Stress and Coping (Lazarus & Folkman, 1984) is a commonly-
cited model for understanding how stress and coping are mediated by cognitive appraisal of an
event. The model emphasises how appraisal of an event will determine how the event is
psychologically managed, and whether coping resources are required to manage the event. For
parents experiencing paediatric chronic illness, appraisals of a child’s conditions set in motion any
number of emotion-focussed and problem-focussed coping strategies. How parents cope and what
strategies and styles of coping they use are important explorative components of my research.
Consequently, providing comprehensive discussion about the Stress and Coping Model has enabled
a detailed description of specific aspects of the Resiliency Model to which my thesis pays particular
attention. Within the description of the Stress and Coping Model, the framework of appraisals and
coping strategies may be relevant to findings of coping mechanisms identified in my thesis.
The Resiliency Model of Stress, Adjustment and Adaptation (McCubbin & McCubbin, 1993) is
a theoretical model developed with families who have a child with CF. The Resiliency Model
represents the shift both from an individual and psychopathological view of parenting a child with a
chronic condition to a holistic, strength-based approach to understanding how parents manage their
child’s chronic condition. The framework emphasises resilient properties of families, along with their
strengths and capabilities to manage their challenging situation (McCubbin, et al., 1996). Exploring
how parents and families manage paediatric chronic conditions from a resiliency perspective affords
an opportunity to understand how concepts and their relationships might contribute to
development of cognitive constructions and attributions of meaning that might influence coping and
adaptation. More importantly, because of its emphasis on adaptation to chronic illness, the most
desired response for families experiencing chronic illness, the model’s concepts and relationships
bear relevance to my research questions and objectives. Due to its focus on family processes within
the context of chronic illness and its potential to isolate parental experiences and outcomes, The
Resiliency Model has been chosen as the structure through which parental processes and constructs
identified in my research findings are viewed and interpreted. Discussion will now shift focus to
substantiating paradigmatic and methodological choices for conducting my research
3.3 Study design
3.3.1 Conceptualising qualitative research.
Methodology refers to the way issues are approached and how research is conducted
according to pre-determined assumptions that shape the methods chosen to answer questions
relevant to the identified issue (Gelo Braakmann & Benetka, 2008; Taylor & Bogdan, 1998). Within
psychology methodology, traditionally there are two distinct approaches about which there has
64 Parenting Children with Cystic Fibrosis
been much debate regarding the criteria for knowledge and the subsequent decisions in reference
to appropriate methodological decisions (Corbin & Strauss, 2008; Gelo et al., 2008; Majima &
Moore, 2009). Firstly, quantitative research refers primarily to positivist claims for developing
knowledge whereby strategies of inquiry include experiments and surveys using predetermined
instruments, which yield statistical data. Secondly, a qualitative approach to research refers
primarily to constructivist perspectives utilising strategies of inquiry such as narratives and
phenomenology to collect open-ended, emerging data with the primary intent of developing themes
from the data (Creswell, 2014). Therefore, despite the debate surrounding the decisions and use of
quantitative or qualitative methodology, it is now widely accepted that the decision around
methodological preference should be based on adopting a method appropriate to investigate the
research question (Chamberlain, 2000; Corbin & Strauss, 2008; Gelo etal., 2008; Grbich, 2007;
Liamputtong, 2009; Vlasiner, 2006).
A simplistic explanation of the primary difference between qualitative and quantitative
research is that studies employing a qualitative form of methodology rely on linguistic rather than
numerical data, and adopt meaning-based rather than statistical forms of data analysis (Elliott &
Timulak, 2005). Other distinctive features of qualitative research include: an attention to
understanding phenomena in their own right (as opposed to an outside perspective); open and
exploratory research questions (versus closed-ended hypotheses); unlimited and emergent
descriptive options (rather than predetermined choices of rating scales); use of specialised strategies
for strengthening credibility of design and analyses; and definition of success in terms of discovering
something new (versus confirming what was hypothesised) (Elliott, Fischer, & Rennie, 1999).
Qualitative research is inquiry aimed at describing and clarifying human experience as it
appears in people’s lives (Polkinghorne, 2005). Qualitative methodology provides an essential quality
of data through well-grounded, rich descriptions and explanations of processes occurring within
populations (Miles & Hubermann, 1994). Techniques such as interviews allow researchers to acquire
knowledge and an understanding of issues for a small sample of participants in much more depth
(Patton, 2002). This methodology is the most appropriate when the researcher is attempting to
understand unexplored complex values, emotions or ideas. Due to the exploratory nature and
purpose of qualitative research, data analysis is an inductive (rather than deductive) process, and
exploratory rather than confirmatory. The qualitative data analysis process is discussed in detail
further in the chapter.
The qualitative research approach has its roots in both cultural anthropology and American
sociology (Kirk & Miller, 1986). The intention is to understand a particular situation, event, role,
group or interaction through an investigative process whereby the researcher makes sense of a
social phenomenon by contrasting, comparing, replicating, cataloguing and classifying the object of
65 Parenting Children with Cystic Fibrosis
investigation (Miles & Huberman, 1994). This intense investigation requires immersion in the
everyday life of the population chosen for the study. The researcher enters the participants’ worlds
and through ongoing interaction, seeks participants’ perspectives and meanings (Marshall &
Rossman, 2010).
Qualitative research has traditionally been used in research disciplines such as anthropology,
sociology and psychology, but rigorous qualitative research methods over the past 20 years have
resulted in a surge of this type of research in social science and increasingly in the health sciences,
particularly nursing (Elliott, et al., 1999; Polit & Beck, 2014). Historically, the definition of qualitative
methodology has evolved over time (Creswell, 2007; Denzin & Lincoln, 1994, 2000, 2005) to include
an emphasis on the process of research, that is, as continuing from philosophical and theoretical
assumptions (Creswell, 2007; Denzin & Lincoln, 2008).
Most qualitative research assumes that to truly and holistically understand people’s
experiences, researchers must attempt to understand the meanings and interpretations that people
give to their experiences. That is, to “uncover the essential structure of reality” (Crotty, 1996, pp.
68). This means then that qualitative methods provide an insight into how people make sense of
their experience that cannot be easily provided by other methods (Liamputtong & Ezzy, 2005).
Typically, qualitative research has a deep involvement in exploratory questions, and is
suitable as the basis for inquiry when existing research in an area is contradictory or confusing, when
the topic under investigation is complex (Elliot & Timulak, 2005) or when there is a lack of research
in a particular area (McDonald & Daly, 1992). It has also been suggested by Holman (1993) that
qualitative research is especially insightful, and can greatly contribute to the study of chronic illness
because, as he suggests, patients’ views are “better indicators of outcomes than common medical
measures” (p. 33).
Therefore, to appropriately address these types of issues, open-ended research questions
that guide participants and direct the research as data are derived from their own lived experiences
are necessary (Corbin & Strauss, 2008). This means that the explorative nature of qualitative inquiry
is particularly useful for gaining a holistic understanding of experiences, with emphasis placed on the
understanding of relationships within systems and the on the understanding of processes involved in
meaning making (Corbin & Strauss, 2008; Creswell, 2007; Janesick, 2000). Qualitative inquiry allows
the researcher to create the essence of the experience to determine how meanings of the
phenomena are formed (Denzin & Lincoln, 2008). Phenomena are described by participants and
studied by the researcher (Crotty, 1996). A qualitative methodology is supportive of the
philosophical position of a phenomenological and constructivist approach to research, which will be
explained.
66 Parenting Children with Cystic Fibrosis
The focal point of qualitative research is gaining an in-depth and comprehensive
understanding of particular individuals or groups, rather than investigating the general
characteristics of a large population of individuals across particular variables (Polgar & Thomas,
2008). In contrast to the linear progression of tasks in quantitative research, qualitative research is
more reiterative with the progression of the research following more of a circular motion. This refers
to the flexible and reiterative construction of, and actions within, the study, inclusive of tasks such as
sampling, collecting, analysing and interpreting data (Polit & Beck, 2014).
In qualitative research, no attempt is made by the researcher to control for extraneous
variables, and operational definitions are not provided for the study variables. Rather, human
experiences under investigation are perceived and described as a whole within their particular
context (Polgar & Thomas, 2008). Having chosen the type of qualitative research to be conducted,
specific strategies of inquiry exist that will guide subsequent research processes including data
collection, analysis and writing (Creswell, 2003).
The collection of data usually involves speaking with those who have first-hand experience
and generally consists of loose discussions between the researcher and the participant about the
phenomena under investigation. Loosely structured discussions allow participants to express a range
of values, beliefs, emotion and behaviours associated with the phenomena under investigation. Data
analysis and interpretation are concurrent activities that consist of clustering related narrative
information into a coherent format. Through a process of inductive reasoning, the researcher begins
to identify themes that are used to build a rich description of the phenomena (Polit & Beck, 2014).
Therefore, qualitative research adopts an interpretative, holistic approach to the
phenomena under investigation and involves making sense of the subject matter through
interpreting meanings that participants give to them (Denzin & Lincoln, 2008; Patton 2002). Relevant
to my research, it has been suggested that qualitative research assists to shape and give life to
clinicians’, nurses’ and other allied health professionals’ perceptions of issues or situations, their
conceptualisations of potential solutions, and their understanding of patients’ and families’ concerns
and experiences (Polit & Beck, 2014).
3.3.1.1 Methodological and theoretical perspectives and implications.
Accepted paradigms within qualitative methodology include post-positvism, critical theory
and interpretivism (Denzin & Lincoln, 2003; Wills, 2007). A distinction between the two
methodological approaches of quantitative and qualitative research is the interpretative nature of
the qualitative research process. Consideration of interpretation is apparent throughout the entire
research process, from acknowledging the researcher’s own historical context at each stage of the
research to defining outcomes through an interpretative process of co-construction of reality
between researcher and participants.
67 Parenting Children with Cystic Fibrosis
Therefore, an interpretivist paradigm has the assumptions of a constructivist epistemology.
Constructions of knowledge are conditioned by experience and culture and are therefore context
and perception driven (Wills, 2007), and understanding of the social world is achieved through
exploring the interpretation of that world by its participants (Bryman, 2012). Accordingly, the
knowledge of reality is not available solely through direct experience, rather it is thinking and
reflecting that are important facets of the process of knowing and understanding (Wills, 2007). This
concept and its implications will be discussed in detail in the next chapter.
The broad notion of qualitative methodology is further broken down to specific types of
methodology, for example, grounded theory, ethnography, discourse analysis, heuristic inquiry and
phenomenology (Crotty, 1998; Elliott & Timulak, 2005). Concerning the decision of appropriate
methodological frameworks and theoretical perspectives in view of the world as interactions
between people and people, and people and meaning, there is a requirement of multiple
perspectives on issues and acknowledgement of human experience in broader events situated in a
social, cultural and political framework (Corbin & Strauss, 2008; Gergen 1985). To reveal the
complexity and layering of the phenomenon under investigation in my research, an underlying
theoretical framework informed this research, namely phenomenological inquiry. The theoretical
foundation of this framework is discussed in detail in the next section with regards to how the
position informed and directed my research.
3.3.2 Qualitative methods.
A qualitative approach to inquiry was used in my research as it represented the most
appropriate way to investigate the overarching research questions (Corbin & Strauss, 2008;
Liamputtong & Ezzy, 2005). When little or no previous knowledge has been generated about a
particular topic or within a particular population, and one is trying to understand intrinsic
experiences of individuals to learn how meaning is constructed, a qualitative approach is appropriate
(Creswell, 2007; Liamputtong, 2009). Furthermore, a phenomenological methodological approach is
supportive of the philosophical position of constructivism within the setting of interest.
Phenomenological methodology utilises thick descriptions of a lived experience to explain
different phenomena (Creswell, Hanson, Plano & Morales, 2007; Martin, 2000). Data can be
collected from a variety of empirical materials (for example, interviews), which describe moments
and meanings in people’s lives with a range of interconnected, interpretive processes used to
acquire a deeper understanding of the phenomena under investigation (Denzin & Lincoln, 2008). The
result of this is rich contextualised descriptions of categories that provide an in-depth understanding
of the concepts of interest, within the context in which they occur (Liamputtong & Ezzy, 2005).
These rich descriptions can help to explain why a particular phenomenon occurs rather than simply
learning what other variables relate to the phenomena or how frequently it occurs.
68 Parenting Children with Cystic Fibrosis
Common characteristics of a qualitative methodological design, and more specifically
phenomenology, include: the use of a natural setting for data collection (that is, for example, an
interview conducted in the participant’s home); acknowledgment of the researcher as a key
instrument in the research process (that is, researcher develops the interview schedule, collects and
interprets the data); and the use of inductive data analysis techniques (that is, a reiterative process
to build themes). An inductive design is advantageous when little or no prior research has been
conducted, or when there is a trend towards using quantitative methodology, because it creates an
opportunity to develop new theory and be open to participant meanings. Utilisation of inductive
design means that the researcher is not tightly prescribed to research processes, and this allows
alternative ideas to emerge throughout the research that may be different from those the
researcher brings to the process (Creswell, 2007; Liamputtong & Ezzy, 2005; Miles & Hubermann,
1994; Silverman, 2004). By using interviews as a vehicle for data collection, the researcher can reach
areas of reality that would otherwise remain inaccessible through quantitative investigation, such as
people’s subjective attitudes and experiences (Perakyla & Ruusuvuori, 2011). Analyses of data are
then presented based on participants’ perspectives as well as on the researcher’s own
interpretations (Creswell, 2007).
A key criticism of qualitative research is that it does not account for considerations of
reliability and validity, and provides no account of how data were analysed or interpreted (Johnson
& Onwuegbuzie, 2004). However, this criticism has been addressed in my research by adhering to
strategies of rigour suggested by Guba and Lincoln (1994). The next section will outline Guba and
Lincoln strategies used to achieve scientific rigour. The next chapter will elaborate on how these
strategies were achieved in my research.
3.3.2.1 Scientific rigour.
The quality of all types of research depends on the quality of the methods used (Mays &
Pope, 2000). It has been argued that evaluation of quality in qualitative research by the same
measures and criteria used to assess the quality of quantitative research is fundamentally and
epistemologically flawed (Hammersley, 2007; Kitto, Chesters & Grbich, 2008; Lincoln & Guba, 2000;
Morrow, 2005). The research frameworks, data collection, reliability and validity processes involved
in quantitative research do not fit the qualitative paradigm.
In qualitative research, rigour is the concept for quality of data and integrity of the findings;
that the researcher can demonstrate the findings are worth paying attention to and worth taking
into account (Lincoln & Guba, 1985). Rigour in qualitative research is relevant to the notions of
validity and reliability in quantitative research, and to the reduction of bias. In qualitative research,
validity does not carry with it the same meaning as it does in quantitative research, nor is it a
69 Parenting Children with Cystic Fibrosis
companion of reliability or generalisability (Creswell, 2003). Therefore, the concepts of reliability and
validity need to be reconceptualised to suit qualitative methods.
Validity in qualitative research is used to determine whether the findings are an accurate
representation from the view of the researcher, participant and the readers of an account (Creswell
& Miller, 2000). Terminology abounds in the qualitative literature that speaks of this idea, terms
such as rigour, trustworthiness, authenticity and credibility (Creswell & Miller, 2000), and is a highly
debated topic (Lincoln & Guba, 2000). Lincoln and Guba (1985) proposed that constructivist inquiry
required a different set of criteria for assessing quality from those of traditional social sciences;
collectively the criteria are an indication of trustworthiness, a parallel to the term rigour. There are
several elements of rigour within the research process that need to be achieved.
3.3.2.1.1 Operationalising rigour in qualitative research.
Viewed as the gold standard for rigourous qualitative research, Lincoln and Guba’s (1985)
framework of quality criteria suggests four criteria for developing rigour and trustworthiness in
qualitative research; credibility, dependability, confirmability, and transferability. These criteria
represent parallels to the positivists’ criteria of internal validity, reliability, objectivity, and
generalisability, respectively (Polit & Beck, 2014). In response to criticisms and to their own evolving
views, Guba and Lincoln (1994) added a fifth criterion more distinctively within the constructivist
paradigm; authenticity.
Credibility is the most important measure of rigour, and refers to establishment of
confidence in the truth value of the data, and the resultant interpretative findings (Lincoln and Guba,
1985). Two aspects of qualitative research are essential to achieving credibility; conducting research
in such a way that enhances believability of findings; and implementing processes that demonstrate
credibility to individuals external to the research. Its quantitative equivalent is internal validity,
which when adapted and applied to the qualitative paradigm ensures that the findings represent the
multiple constructions of an experience adequately; that the reconstructions that have been arrived
at are credible.
Dependability refers to stability, or reliability, of data over time and across contexts. This is
achieved through replication of findings with the same (or similar) participants in the same (or
similar) context. Credibility cannot be achieved in the absence of dependability, just as validity
cannot be achieved in the absence of reliability in quantitative research (Lincoln & Guba, 1985).
Confirmability is the potential for congruence between two or more independent people
about the data’s accuracy, relevance and meaning. This element of rigour is concerned with
establishing that the information participants provide is representative of the data, and that
interpretations of data are not created by the researcher. This is achieved by displaying that the
70 Parenting Children with Cystic Fibrosis
findings reflect participants’ voices and the conditions of the research, and are not the product of
the researcher’s motivations, perspectives, or biases. Confirmability relates to the concept of
objectivity because it demonstrates where and how the findings of the research are derived from
the participants and conditions of the research and not by the biases, motivations, or perspectives of
the researcher (Lincoln & Guba, 1985).
Transferability, analogous to generalisability, is the extent to which the findings can be
transferred, or apply to, other groups or settings, including provision of sufficient descriptive data so
that others can evaluate the applicability of the data to other contexts. Generalisability is impossible
to achieve and does not suit the framework or aims of qualitative research. The qualitative
researcher can only provide sufficient description for consumers to reach their own conclusions as to
whether findings are applicable and transferable to other contexts (Lincoln & Guba, 1985).
Authenticity is the extent to which researchers fairly and faithfully show a range of different
realities. This emerges in a report when it conveys the tone of participants’ lives as they are lived. A
study has authenticity if it leads the reader into a vicarious experience of those who are being
studied and provides readers with a heightened sensitivity to the experiences, and issues,
characterised.
In summary, a qualitative methodology adopts an interpretive, naturalistic approach to the
phenomenon of interest and involves making sense of a phenomenon through interpreting
meanings that participants construct about them (Denzin & Lincoln, 2008; Patton, 2002). My
research employed a qualitative research methodology within a phenomenological framework using
in-depth exploratory interviews with a purposive sample of participants, to address the research
questions of what are parents’ lived experiences and which factors are important in understanding
constructions of experiences and attributions of meaning for parents whose children are involved in
the AREST CF early surveillance program, and how parents cope. The qualitative framework sought
to explore and describe the experiences, thoughts, emotions and ideas of participants by co-
constructing the different ways participants experience parenting in the context of early surveillance
for lung disease, and the meanings associated with those experiences. Explanation of
phenomenology as the underpinning principles of the chosen methods will now be explained before
providing justification of data collection, sampling and data analysis techniques will now be outlined
before detailing why, with regard to all the reviewed literature, my research addressed a pressing
lack of understanding, and was therefore worthy of action.
3.3.2.1 Phenomenology.
Phenomenological theory has influenced qualitative research, in particular in the social
sciences (Flood, 2010; Liamputtong & Ezzy, 2005; Zippel, 2010). It is popular in the health sciences
(Borgatta & Borgatta, 1992), psychology (Giorgi, 1985, Polkinghorne, 1989), and nursing
71 Parenting Children with Cystic Fibrosis
(Nieswiadomy, 1993). Phenomenology is derived from philosophy and provides a framework for a
method of research (Mapp, 2008). As the philosophical underpinnings of phenomenological inquiry
direct a phenomenological study, it is imperative to understand how one relates to the other. A
phenomenological approach focuses on the lived experiences of people and seeks to understand
their reality from their perspective, emphasising the direct study of personal experience and
understanding of the nature of human consciousness (Becker, 1992). Phenomenology is based
within an interpretivist research paradigm and follows a qualitative approach (Denscombe, 2003).
These concepts, fundamental to understandings of phenomenological inquiry and to qualitative
research practice, have developed over time through contributions from influential
phenomenological philosophers (Flood, 2010; Liamputtong & Ezzy, 2005; Patton, 2002).
Phenomenology as a philosophical method of inquiry was developed by the German
philosopher, Edmund Husserl (1859-1938), who is credited with the birth of phenomenology and
developed a systematic method of studying human consciousness and experience (Husserl, 1977).
Husserl posited that experiences of life events in the everyday world, with theoretical underpinnings
suspended, was an invaluable source of knowledge (Becker, 1992). His aim was to return to things
themselves, as they appear to their perceivers, and to set aside, or bracket, that which we (think)
already know about them (Willig, 2001). Husserl developed phenomenological inquiry to study how
people describe things, with the assumption that we can only know what we experience by
attending to meanings that activate conscious awareness (Flood, 2010; Patton, 2002). Husserl
referred to this notion of conscious awareness of how experiential processes proceed and what is
experienced through them as intentionality (Charmaz & McMullen, 2011). Therefore, people
construct meanings as they engage with, and make sense of, the world they are interpreting. It is the
researcher’s task to analyse the intentional experiences of consciousness to determine how
phenomena are given meaning and to arrive at its essence (Sadala & Adorno Rde, 2002). An analysis
of the essence (or structure) of phenomena in context is one of the outcomes of phenomenological
research (Flood, 2010).
Another German philosopher, a student of Husserl’s, Martin Heidegger (1889-1976),
continued Husserl’s works by including elements of space and time within this broader explication of
phenomenology (1962). Heidegger’s work is cited as the first union of existentialism and
phenomenology, resulting in existential-phenomenology; a study of human existence and
experience with existential foundations within a phenomenological method (Becker, 1992). Whilst
Heideggerian phenomenology proposes that researchers interpret data in terms of their own
experiences and knowledge, Husserlian phenomenology advocates that researchers suspend (or
bracket) their personal beliefs about the research phenomena whilst seeking to describe the
participants’ experiences (Mapp, 2008).
72 Parenting Children with Cystic Fibrosis
More recently, phenomenological inquirers have shifted from a pure philosophical method
of inquiry to adapting and systematising the use of phenomenological methods. Led by Amedeo
Giorgi (1985, 2009), methods originally developed by phenomenological philosophers, such as the
intentional analyses of meaning and the analysis of the essences of phenomena have been adapted
for empirically based psychological research (Wertz, et al., 2011).
Despite differences of opinion amongst qualitative researchers who use phenomenological
inquiry, there are a number of commonalities in its philosophical beliefs. These include the study of
lived experiences of a group of people who experience a particular phenomenon and the
consideration of these experiences as being conscious ones (Flood, 2010; van Manen, 1990; Zippel,
2010), and the process of developing a description of the essence of these experiences (Flood, 2010;
van Manen, 1990). At a broader level of commonality is the rejection of a subject – object
dichotomy, therefore assuming that reality of an object is perceived only within the meaning of the
experience of an individual (Creswell, 2007; Flood, 2010; Zippel, 2010). In considering these
commonalities, phenomenological inquiry is concerned with co-construction of everyday life
emphasising that people’s actions are understood only when they are situated in the routines of
everyday interactions in the world (Flood, 2010; Liamputtong, 2009). Therefore, a phenomenological
study seeks to explore and describe people’s experience of everyday life events within a definite
philosophical context that produces specific assumptions about human nature and humans living
within a particular nature (Becker, 1992).
Accordingly, phenomenological inquiry in philosophical terms asserts the nature of how
reality comes to be experienced and understood (Flood, 2010; Grbich, 2007; Liamputtong, 2009;
Patton, 1990), whereas as a research methodology, phenomenological inquiry seeks to explore how
a specific aspect of lived reality is constructed by participants. This includes, on the researcher’s
behalf, bracketing preconceived ideas about the phenomena in question (Daly, et al., 2007;
Carpenter & Suto, 2008). As either a philosophical approach to research or a methodology, its
central focus is about how to investigate the lived experience of a particular phenomenon for those
who experience it (Creswell, 2007; Liamputtong, 2009; Moustakas, 1994; Patton, 1990). As a
research methodology for understanding the lived experiences of individuals (as in the work of
Giorgi, 1985, Giorgi and Giorgi, 2003), phenomenology is a popular framework within the social and
health sciences, particularly in sociology, psychology, education, nursing and health sciences
(Creswell, 2007; Wills, 2007; Padgett, 2008).
When using phenomenology as a methodological framework, a study aims to understand
and describe participants’ experiences of their everyday lifeworld as they see it (Wills, 2007;
Carpenter & Suto, 2008; Padgett, 2008). This understanding of phenomenological inquiry aligns with
the purposes of my research, which seeks to explore parents’ lived experiences of early surveillance
73 Parenting Children with Cystic Fibrosis
and interpret how meaning is assigned (Creswell, 2007; Flood, 2010). For this reason, the
significance of phenomenological inquiry to my research is in gaining an understanding of how
parents experience the phenomena of early surveillance. Of particular concern is how parents’
interactions in the world influence their experiences. As such, how parents construct their
experiences becomes a focal point. Accordingly, the philosophical underpinnings of a constructivist
approach to research are discussed in the next chapter.
In terms of practical application, a phenomenological approach to understanding experience
has led to positive health outcomes. Some examples of where phenomenological enquiry has led to
positive health outcomes from knowledge gained in nursing research include: phenomenological
analysis of interviews with patients who suffered from obstructive sleep apnea enabled
development of a tailored intervention that promoted treatment adherence (Sawyer, Deatrick, Kuna
& Weaver, 2010); a psychoeducation program was developed from phenomenological methodology
that improving physical functioning, general health, anginal pain symptoms, and self-efficacy to
manage pain (MCGillion, et al., 2008); and phenomenological analysis allowed elaboration of
quantitative findings that showed anxiety and anger increase children’s blood pressure.
Phenomenological enquiry in this particular study discovered what aspects of childhood can
contribute to anxiety and anger, which contributed to development of a program for teaching
children how to deal with their emotions (Howell, Rice, Carmon & Hauber, 2007).
As there are no studies exploring parents’ experiences of early surveillance for CF lung
disease, a phenomenological study is appropriate for eliciting data that reflect parents’ experiences
and the ways in which they interpret, and cope with, early surveillance. Therefore, in the context of
my research, I viewed experience as a valid and worthwhile source of knowledge, and everyday
worlds of individuals as also valuable sources of knowledge. Accordingly, a phenomenological study
exploring parents’ experiences of caring for a child being treated for CF enrolled in the AREST CF
early surveillance program was conducted.
3.3.2.3 Data collection technique.
There are a number of informative sources and data collection methods that can be used in
qualitative research (Chwalisz, Shah & Hand, 2008; Patton, 2002). A key strategy is to obtain
accounts or descriptions of the phenomena under investigation through conversations with people,
and to learn about their lived experiences or their life world (Creswell, 2007; Kvale & Brinkmann,
2009). Techniques such as narrative interviewing, conversational interviewing and semi-structured
interviewing attempt to understand the world from the participant’s perspective and aim to uncover
meanings of their experiences, rather than to provide scientific explanations (Kvale & Brinkmann,
2009, Liamputtong & Ezzy, 2005; Wengraf, 2001). Moreover, it is assumed that semi-structured
interviewing techniques elicit specific knowledge, emotions, perceptions and experiences of a
74 Parenting Children with Cystic Fibrosis
phenomenon that can be articulated best by the verbal accounts of participants in their own words
(Liamputtong, 2009). For this reason, a semi-structured interviewing technique is an appropriate
approach to investigate the essence of the lived experiences of participants in my study (Creswell,
2007; Elliott & Timulak, 2005).
A semi-structured interview schedule consists of a set of broad questions about a topic that
allows freedom in conversation (Liamputtong, 2009; Patton, 2002). This method was chosen as the
vehicle for data collection because it allowed me to adequately explore parents’ experiences whilst
at the same time allowing substantial flexibility in conversation between participants. It also allowed
the use of probing questions to develop more layered responses and descriptions that may be of
particular interest or relevance (Chwalisz, et al., 2008; Elliot & Timulak, 2005).
The flexibility afforded by the semi-structured interview is particularly appropriate to
discovering a multitude of, and complexity within and across, experiences and associated meanings.
Miles and Hubermann (1994) and also Smith (1995) identified several advantages of conducting
semi-structured interviews, which include assurance of flexibility in developing rapport, ensuring
suitability to exploring personal and complex issues, and in providing rich data embedded in a
certain context. Additionally, this style of interview suits a constructivist framework because it frees
the researcher to explore how people construct meanings about their experiences, which leads to
obtaining rich descriptions of the life world of participants (Liamputtong & Ezzy, 2005), and also
applies to the consideration that one real truth does not exist in place of multiple realities (Guba &
Lincoln, 1982).
The nature of this qualitative research project required that I have an intuitive sense of what
was occurring in the data; therefore flexibility was instrumental throughout the entire research
process (Corbin & Strauss, 2008). Flexibility in the semi-structured interview format allowed parents’
experiences and stories to guide the interview, which meant they became active participants in the
research process rather than respondents to a fixed set of questions (Elliot & Timulak, 2005;
Polkinghorne, 2005). Gaining knowledge and understanding of participants’ experience in such a way
provides greater detail and allows for consideration of the wider context of experience that is
important when dealing with complex emotions and values about a phenomenon (Miles &
Hubermann, 1994; Patton, 1990).
Indicative of the dynamic flexibility required within a qualitative research design, it become
apparent during data collection that telephone interviewing could increase volume of participation,
therefore increasing the diversity and amount of parental voices heard. Telephone interviewing has
been used extensively in quantitative research methods (Barriball, Christian, While & Bergen, 1996;
Carr & Worth, 2001), mostly as a form of survey data collection. However, this particular interview
mode has been much less used as a qualitative method (Sturges & Hanrahan, 2004), with debate
75 Parenting Children with Cystic Fibrosis
about its adequacy (Carr & Worth, 2001; McCoyd & Kerson, 2006; Novick, 2008; Sturges and
Hanrahan, 2004; Sweet, 2002). As a method of data collection, telephone interviews possess several
advantages including increased access to geographically distant participants (Sturges and Hanrahan,
2004; Sweet, 2002), the ability to take notes unobtrusively (Carr & Worth, 2001; Sturges and
Hanrahan, 2004), more privacy (Sturges and Hanrahan, 2004), increased rapport (McCoyd & Kerson,
2006), convenience to participants, and elimination of any influence to respondent replies by
characteristics of the interviewer, for example, class or ethnicity (Bryman, 2006). Although face-to-
face interviews appear in the literature to be the ‘gold standard’ for qualitative research (McCoyd &
Kerson, 2006), the use of telephone interviews has been reported as a versatile data collection tool
(Carr & Worth, 2001), with telephone interview data judged as a source of rich, vivid, and detailed,
high quality data (Chapple, 1999; Kavanaugh and Ayres, 1998; Sturges and Hanrahan, 2004; Sweet,
2002).
In my research, I was required to identify and acknowledge myself as part of the research
instrument and to view the research as transitional and malleable and therefore open to
modification and refinement as new knowledge was gathered (Corbin & Strauss, 2008). I engaged in
a process of reflexivity, which involved continual and detailed self-reflection (see in later sections:
Role of the Researcher and Rigour in Qualitative Research). The interpretative nature of this
research, and my continual and intricate involvement in the research process, produced a range of
strategic, ethical and personal issues in the research process (Locke & Golden-Biddle, 2002).
In summary, qualitative methodology and semi-structured interviews with parents were
used to explore, understand and interpret the lived experience of parenting children with CF in the
context of early surveillance. Employing a qualitative methodology allowed me to move away from
quantification and causation towards discovering the essence and meaning attributed to these
experiences. The style of the qualitative interview allowed me to observe experiences relative to a
person’s construction of phenomena and it’s relation to their life world. Obtaining an inclusive
account of parents’ experiences was paramount to interpreting meaning of the described
phenomena (Kvale & Brinkmann, 2009; Liamputtong, 2009; Polkinghorne, 2005). The next section
outlines the chosen sampling technique.
3.3.2.4 Sampling technique.
A primary aim of qualitative research is to derive meaning, to uncover multiple realities, and
to avoid generalisation to a target population (Creswell, 2014). Therefore, qualitative sampling
completely avoids random sampling as it is not the most appropriate method for selecting
individuals who will be useful informants. Non-random sampling identifies participants who have the
relevant experience to meet the conceptual needs of the study and provide the information sought.
Therefore, qualitative researchers use non-probability sampling techniques (Polkinghorne, 2005).
76 Parenting Children with Cystic Fibrosis
Fundamental to understanding the experience of individuals is to question those who have
the relevant experience (Seidman, 1989): to understand what it is like for them and from their
perspective (Dewey, 1960). Accordingly, my research applied a purposive sampling technique, which
involved selecting participants, from a cohort of potential cases, who would best contribute to the
research (Creswell, 2014). Maximum variation sampling (Patton, 2002) was chosen as the specific
type of purposive sampling as it engages cases with demographic or other differences that have
shared a common experience. This type of purposive sampling is often the sampling technique of
choice in qualitative research because it is useful in exploring a broad scope of the phenomena and
in identifying critical patterns that cut across variations in demographics or other differences
identified through the sampling technique (Polit & Beck, 2014). Qualitative researchers often want
to explore and understand the breadth of individual experience and so specifically look for
individuals with demographic or other differences who have shared a common experience (Seidman,
1989).
My research recruited parents who have experience of their young child with CF enrolled in
the AREST CF early surveillance program. To achieve the breadth of parental experiences, both
mothers and fathers of children who attend the CF clinics at PMH, Western Australia and RCH,
Victoria were invited to participate. The sample was stratified at recruitment into two groups
according to the child’s age (under 3 years and over 3 years). This allowed representation of
maternal and paternal experiences across time in the programme, as well as sex-specific and clinic
site-specific issues.
The purpose of stratified-purposive sampling (Patton, 2002) is to identify major variations in
experience rather than to identify a common core experience. Stratification by child age provided
some representation of the child’s stage of development in relation to parents’ experiences and
ability to cope, and captured potential themes specific to the duration of time the child has received
early surveillance since diagnosis.
3.3.2.5 Sample size and saturation.
In qualitative research, no set procedure is strictly applied to determine the sample size, as
is the case for quantitative research (Patton, 2002).The purpose of sampling in qualitative research is
to focus in-depth on relatively small samples selected meaningfully and strategically, rather than for
the purpose of attempting to make statistical comparisons (Patton, 2002). Therefore, the sampling
process is flexible and at the start of the study the number of participants is not definitively known.
Data saturation, however, is a procedure used by qualitative researchers as a way of justifying the
number of participants; this occurs during the data collection process (Liamputtong, 2009).
Saturation occurs when no new data are being generated from the data already collected
(Padgett, 2008) and sufficient data have been obtained to account for all aspects of the
77 Parenting Children with Cystic Fibrosis
phenomenon (Morse, Barrett, Mayan, Olson, & Spiers, 2008). This refers to the idea that the sample
is adequate when “the emerging themes have been efficiently and effectively saturated with optimal
quality data” (Carpenter & Suto, 2008, p. 152). Mason (2010) and Morse (2000) both report
adequate saturation with approximately 30 interviews, with Francis and colleagues (2009), Guest
and colleagues (2006) and Burgess-Limerick and Burgess-Limerick (1998) all suggesting 12-15 in-
depth interviews could be adequate.
3.3.2.6 Thematic analysis.
Engagement in exploratory qualitative research relating to parents’ constructions and
experiences of parenting children with CF in the context of early surveillance relies on inductive
processes such as thematic analysis whereby themes emerge from the data that provide rich,
context-bound information (Creswell, 1994, Kvale & Brinkmann, 2009). Coding is the primary process
for developing themes within the raw data by recognising important moments in the data and
encoding it prior to interpretation (Boyatzis, 1998). Given thematic analysis is an analysis tool for
exploring interview data, it allows identification, examination and reporting of themes within data
and provides foundations for describing rich information (Braun & Clarke, 2006; Kvale & Brinkmann,
2009; Liamputtong, 2009).
Outcomes of thematic analysis are called themes, and are considered representative of a
level of patterned response or meaning or construction that attempt to address the research aims
and questions (Braun & Clarke, 2006). Though description is part of the analytic journey of which
thick description is a valuable component, description alone is not sufficient. The data must also be
challenged, extended, supported and linked to reveal their full value. Themes only attain their full
significance when they are linked to form a coordinated picture or an explanatory model (Bazely,
2009). As the theoretical framework of phenomenology and philosophical assumptions of
constructivism guided this research, the ways in which individuals make meaning and construct
experience were of particular interest. Accordingly, thematic analysis offers a theoretically flexible
approach to analysing qualitative data (Braun & Clarke, 2006; Elliot & Timulak, 2005) because it
allows the researcher to organise qualitative data coherently (Miles & Hubermann, 2002). Braun and
Clark’s (2006) stage process for thematic analysis aligns with Giorgi’s (1985) methods of
phenomenological analysis.
3.4 Summary
This section has shown how the epistemological and methodological assumptions of my
research were conceptualised in response to the phenomena under investigation and the research
questions being explored. This chapter has presented substantiative evidence for theoretical and
methodological choices underpinning my research. Theoretical frameworks provide a structural
guide, whereas methodological strategies provide an operational guide. Together, they function as a
78 Parenting Children with Cystic Fibrosis
basis through which to conduct research, view research findings, and to generate interpretations
and to make recommendations. The next chapter will describe the methods used to conduct my
research.
79 Parenting Children with Cystic Fibrosis
CHAPTER 4
Methods
This chapter presents the methods used to obtain data pertaining to parents’ experiences and
coping strategies, and to interpret which factors are important in understanding the experience of
parents who have a child with CF undergoing early surveillance for lung disease. The purpose of this
chapter is to; 1) reiterate the aims of the study and outline the research questions; 2) review the
theoretical framework used to conduct the study; 3) detail the participants involved; 4) describe the
recruitment process; 5) explain data collection procedures; 6) outline data analysis techniques and
highlight rigourous techniques within a qualitative study and finally; 8) address ethical
considerations and time frame.
80 Parenting Children with Cystic Fibrosis
What we observe is not nature itself,
but nature exposed to our method of questioning...Werner Heisenberg
4.1 Restatement of aims
Despite growth in early surveillance over the past decade, both in terms of the amount of
medical procedures performed on children as well as the recognition of the program in the CF
scientific community, there was a lack of understanding the experiences of parents whose children
undergo early surveillance for CF lung disease. Therefore, the overarching aim of my research was to
explore and interpret the lived experience of parenting in the context of early surveillance for CF
lung disease. More specifically, the exploratory nature of the research set out to identify how
parents construct their experiences and attribute meanings to early surveillance for CF lung disease,
and how they cope.
4.2 Research questions
To address the research questions, an in-depth, interpretive and descriptive qualitative
approach was used to discover and understand the unique experiences of parents in my research.
Specific research questions were explored through a phenomenological framework that sought to
explore and describe the lived experiences, thoughts, emotions and ideas of parents by co-
constructing the different ways they experience parenting children with CF in the context of early
surveillance, and the meanings associated with those experiences. As research questions leading to
qualitative data collection strategies are often open-ended and exploratory in nature (Elliot &
Timulak, 2005), a qualitative methodology from a constructivist perspective was chosen to address
the following research questions:
What is the lived experience of parenting children with CF in the context of early
surveillance?
What are parents’ experiences of early surveillance?
How do parents construct their experience and attribute meanings to early surveillance for
CF lung disease?
How do parents cope with the lived experience of parenting in the context of early
surveillance?
4.3 Methodology
4.3.1 Phenomenology as theoretical framework.
Based on those philosophical influences described in the previous chapter,
phenomenological inquiry as a research tradition intends to describe the life worlds of individuals
(Creswell, 2007; Flood, 2010). Fundamental to phenomenological inquiry is the examination and
description of meaning for a number of individuals about their lived experiences of a particular
81 Parenting Children with Cystic Fibrosis
phenomenon as they are considered to be the best able to describe their experiences (Creswell,
2007; Liamputtong, 2009; Moustakas, 1994; Patton, 2002). Focussing on descriptions of people’s
experiences with the underlying assumption of the existence of a structure (or essence) to shared
experience, central meanings mutually understood through the experience of a phenomenon can be
determined (Moustakas, 1994; Patton, 2002). This description, as previously outlined as
intentionality, includes what and how it is experienced (Creswell, 2007; Moustakas, 1994). This
understanding of the lived experience of individuals is at the heart of phenomenology as a
philosophy as well as a research method (Creswell, 2007; Liamputtong, 2009; Moustakas, 1994).
Therefore, a study with a phenomenological perspective is considered different from using
phenomenology to philosophically justify the methods of qualitative inquiry as legitimate in social
science research (Patton, 2002).
From its philosophical inquisition into the nature of existence to its more practical
application as a method of qualitative inquiry, the evolution of phenomenological inquiry can be
further explained by investigating how data are interpreted. Traditionally, phenomenological inquiry
was viewed as a first-person experience (Husserl, 1977). However, more recently it is common in the
social sciences to observe phenomenological inquiry as a study of other people’s experiences
principally reported in the third person (Heidegger, 1996). Another influential philosopher in the
evolution of phenomenological methodology, Giorgi (1997, 2009) set explicit, systematic and
accountable procedures for analysis of phenomenological data. His methods were in contrast to the
earlier informal way phenomenological research in psychology had been conducted by earlier
pioneers in philosophy, psychiatry and other disciplines. Briefly, the procedures comprise four
stages: reading about the experience for a sense of the whole, differentiating the description into
meaning units, reflecting on the psychological meaning and significance of each meaning unit, and
clarifying the psychological structure (or essence) of the phenomena (Charmaz & McMullen, 2011).
Through reflecting on many manifestations of a phenomenon, I was able to identify common
themes. Moreover, by articulating the inter-relationships of these themes, the essential structure of
the phenomenon was revealed (Becker, 1992). Giorgi’s (1997; 2009) phenomenological data
analytical process is reflected in Braun and Clark’s (2006) explanation of thematic analysis, of which
a detailed discussion can be found further in the chapter.
4.3.2 Constructivism as philosophical underpinnings.
The philosophical underpinnings, or worldview, guiding my research is constructivism. The
purpose of constructivism is to understand how the human mind actively gives meaning and order to
the reality to which it is responding (Balbi, 2008). In contrast to the positivist ontology, which
assumes one reality or truth to be discovered (Guba & Lincoln, 1994), constructivism adheres to a
relativist view of knowledge (ontology), which assumes multiple and equally valid realities
82 Parenting Children with Cystic Fibrosis
(Schwandt, 2000), in that people consciously experience the world and events in diverse ways
(Polgar & Thomas, 2008). This is such that no purely objective knowledge truths are said to exist -
rather, the rules of knowledge are co-constructed and are therefore subject to change.
From an epistemological perspective, constructivism holds that reality is constructed in the
mind of the individual (Hansen, 2004) and co-constructed by researcher and participant. The
constructivist position emphasises that meaning is hidden and must be constructed through deep
reflection (Schwandt, 2000). As such, the nature of this enquiry assumes that knowledge is
constructed – not discovered by the mind, and reality is reflected in a contextualised way (Alvesson
& Sköldberg, 2009, Schwandt, 2000). Therefore, a distinguishing feature of constructivism is the
centrality of the interaction between the researcher and the participants under investigation, which
allows deep reflection to occur through the interactive researcher-participant dialogue (Ponterotto,
2005). It is through this interaction that profound meaning can be understood. This process
therefore allows the joint production (through co-construction) of findings from the interactive
dialogue and interpretation.
Accordingly, based on the interpretivist-constructivist understanding of knowledge, the
epistemological position of knowledge is subjective and influenced by the context of the situation,
including individual experience and perceptions, the social environment and the interaction
between individual and researcher (Ponterotto, 2005). Duly, constructivism claims that reality is
socially constructed and can be understood only within context (Wills, 2007). Thus, context is an
important factor that influences how people interpret their worlds and needs to be considered in
research. This is contrary to positivist philosophy, whereby context is eliminated through using
controls and randomisation, and where gathered research is more representative of experimental
conditions than real life (Guba & Lincoln, 1994).
Within constructive epistemology, researcher and participants are connected in the research
process, findings are created through interaction, and values are acknowledged, whereas within a
positivist epistemology, the researcher remains objective and separate from participants so as to
keep the research unbiased and value free (Charmaz, 2000: Guba & Lincoln, 1994). Consequently, a
researcher in the positivist paradigm becomes disconnected from his or her participants. As the
researcher is viewed as co-constructer of knowledge from a constructivist approach, the axiological
viewpoint maintains that the researcher’s values and lived experience cannot be separated from the
research process (Ponterotto, 2005) and must therefore be acknowledged, described and bracketed.
Bracketing refers to the way in which the intentionality of the researcher can be supressed,
thereby separating usual preconceptions that influence everyday perception so that pure
constituents of conscious experience can be uncovered (Polgar & Thomas, 2008). Intentionality
refers to the human capability for awareness of objects as well as their contextual features, and it
83 Parenting Children with Cystic Fibrosis
allows humans to reason about objects in the world and communicate with others (Polgar &
Thomas, 2008). Bracketing of intentionality, then, is to extinguish prejudices so that the essence of
experience, in relation to the phenomena under investigation, can emerge purely and without bias
or distortion.
Critics of the constructivist approach have questioned its strong emphasis on relativism and
subjectivity (Marshall, Kelder & Perry, 2005; Michael, 1996). The aim of constructivist inquiry is not
to suggest that one absolute truth cannot exist, but rather it is subject to change via the participants’
construction and the researcher’s interpretation. Accordingly, the aim of research underpinned by
such assumptions is to explore and understand how people’s worlds and experiences are
constructed (Chwalisz, et al., 2008; Gergen, 2011; Stenner, 2009), by relying as much as possible on
people’s views of given phenomena whilst at the same time acknowledging meanings are
subjectively constructed (Creswell, 2007). The relationship and interaction between participant and
researcher is significant (Ponterotto, 2005). Retrospective rigour techniques consider how criticisms
and limitations of constructivism may be handled in research, which are discussed at the end of this
chapter. If we are to give parents a voice (Rappaport, 1995) to discuss their experiences of their
child’s CF diagnosis and early surveillance, then it would seem appropriate that a constructivist
perspective be utilised to inform this exploratory operation, especially considering it challenges
assumptions about how meaning is formed (Alvesson & Sköldberg, 2009; Gergen, 2011). This must
be a fundamental consideration within this type of inquiry.
Applying a phenomenological framework from a constructivist perspective and thematic
analysis to my research permits rich description and interpretation of parents’ experiences in the
context of the life-world of the parent, and meaning is attained through construction and attribution
rather than through empirical validity (Alvesson & Sköldberg, 2009; Flood, 2010; Liamputtong &
Ezzy, 2005). Given that methodological procedures within this approach are set in the natural world
(Denzin & Lincoln, 2005), the next section discusses the methods (processes and procedures) and
ethical considerations associated with research set in a natural world context.
4.4 Methods
4.4.1 Sample.
All parents who participated in my research were biological parents of their children who
have CF. Children were selected from AREST CF clinic data bases in Western Australia and Victoria
according to age and participation in early surveillance, and parent details were obtained. Refer to
Figure 2 for a map of Australia where AREST CF operates. All children in the databases were
diagnosed through NBS before 6 months of age. Parents received their child’s CF diagnosis within
the first 3 months after birth.
84 Parenting Children with Cystic Fibrosis
Figure 2. Map of Australia showing the regions where AREST CF operates. Retrieved from www.emapsworld.com
4.4.2 Participant characteristics.
Parents of children living in Australia registered within the AREST CF clinic data bases from
PMH (Western Australia) and RCH (Victoria) CF clinics aged between 6 months and 7 years of age
were invited to take part in my research. Most families lived in the metropolitan area of the major
city within each state, with 17 (25%) parents living rurally across both states. Parents or guardians of
infants and children with an equivocal diagnosis of CF, or those who were not participating in the
early surveillance program were not included. Refer to Table 1 for an overview of how the samples
were recruited.
Table 1. Group descriptions and recruitment details
Participant group
PMH parents RCH parents
Description
Perth AREST CF parents recruited through PMH CF clinic
Melbourne AREST CF parents recruited through RCH CF clinic
Selection method Purposively selected based on their engagement with AREST CF at PMH
Purposively selected based on their engagement with AREST CF at RCH
Number of participants 34 33
85 Parenting Children with Cystic Fibrosis
4.4.2.1 Western Australian sample.
A purposive sample of 34 parents, recruited from a population of N=60 eligible families, of
children aged 6 months to 7 years engaged in the clinical and research aspects of early surveillance
with AREST CF at Perth CF clinic in PMH were interviewed. A total of 24 mothers and 10 fathers
participated in an interview, representing a response rate of 40% and 16% respectively. Seven
interviews were conducted with both mother and father present.
4.4.2.2 Victorian sample.
A purposive sample of 33 parents was recruited from N=71 eligible families, of children aged
6 months to 7 years undergoing early surveillance with AREST CF attending the RCH CF clinic. A total
of 22 mothers and 11 fathers participated in an interview, representing a response rate of 31% and
16% respectively. Another seven interviews were conducted with both mother and father present.
Refer to Table 2 for demographic characteristics of the samples categorised by CF clinic location and
stratified child age.
Table 2. Demographic information of AREST CF parents by CF clinic location and stratified child age
Participant group
PMH parents RCH parents
Variable Under 3 years Over 3 years Under 3 years Over 3 years
Parent (N) 18 16 14 19 Mother 12 12 9 13 Father 6 4 5 6
Age (years) Range 23-52 26-42 24-39 30-43 Mean (SD) 37.13 (6.91) 34.57 (5) 31 (5.27) 33.75 (3.89) Median 37 36 29 33
Age of child Range .8-2.5 3-6.5 1-2.5 4-6 Mean (SD) 1.31 (.52) 4.53 (.99) 1.9 (.41) 5 (.65) Median 1 4 2 5
Number of children Range 1 1-3 1-2 1-2 Mean 0.5 1.5 1.14 1.15
Use of child care Yes 8 9 8 9 No 10 7 6 10
Residential location Metropolitan 16 12 10 12 Rural 2 4 4 7
4.4.3 Recruitment.
The most important people in my research were the participants themselves (Buchanan &
Coulson, 2005), so as part of my role within AREST CF at PMH, rapport with both the clinical staff and
potential participants at PMH was established prior to commencement of my research. This was
86 Parenting Children with Cystic Fibrosis
achieved by spending time in the CF clinic with the CF nurse and also by meeting parents of children
with CF during their routine clinic visits. Once the sample had been identified, I worked alongside the
clinical CF nurse to secure interest in my research.
Eligible participants were identified from each site’s AREST CF database, stored at The
Telethon Kids Institute and RCH respectively. The CF clinical nurse at each site and I worked together
to identify and approach patients’ parents for inclusion in my research. Recruitment was achieved by
distributing an invitation recruitment letter (refer to Appendix A) to eligible families in Western
Australia, accompanied by an information statement (refer to Appendices B and C) outlining the
research and what participation would require. Potential participants were required to make contact
with me, at which time an interview was scheduled for a mutually convenient time and place, or
telephone interviews were suggested as an alternative option when deemed necessary. I
interviewed 50 parents at their homes, with 17 interviews being conducted over the telephone.
Due to differences in geographical location between RCH to PMH (where I am located), the
same process of recruitment and establishing rapport and gaining access to potential participants
was not possible at the Victorian site. The CF clinical nurse coordinator within the Respiratory
Medicine department at RCH assisted in participant recruitment for the Victorian cohort. She
established parents’ interest in my research before granting me permission to contact potential
participants, at which time an invitation letter and information statement were sent. I then made
telephone contact with potential participants to organise an interview time and location before
travelling to Victoria to conduct the interviews, either by telephone or face-to-face.
4.4.5 Ethics considerations.
Ethical issues and dilemmas in qualitative research are emergent and contextual, and
therefore require situational responses. Therefore in keeping with ethics guidelines, it is worthy to
note a number of processes that kept participants informed and safe for the duration of my
research.
4.4.5.1 Research conduct.
All components of the research were conducted in compliance with the relevant codes -
National Statement on Ethical Conduct in Human Research, developed jointly by NHMRC and
Australian Research Council, and the Australian Psychological Society Ethical Guidelines, and in
accordance with my research protocol. Ethics approval for this study was obtained from Edith Cowan
University (8808), PMH (2023/EP) and RCH (33181-A). Research burden was a primary concern as my
study has been with a sample of children with a rare condition requiring specialised care.
Consequently, there has been considerable competition with clinical research groups in gaining
access to the participants. At the time of recruitment, potential participants (and their children) may
have been involved in many different research studies, both clinical and psychosocial in nature.
87 Parenting Children with Cystic Fibrosis
Therefore it was paramount to consider the timing of recruitment, both in Western Australia and
Victoria. By working in conjunction with other AREST CF researchers, it was ensured that my
research did not coincide with other psychosocial research projects.
In terms of participation in my research, I informed parents the interview was confidential,
that they could stop at any time, they could withdraw from the research at any time without
question, that any decision to withdraw would not affect their child’s ongoing CF care, and that they
could ask any questions. They were also advised the interview would be audio-recorded and
transcribed and that a specific code would be recorded on their transcripts and demographic
information to ensure confidentiality. After the research had been comprehensively explained, I
obtained written and informed consent from participants for their involvement and for their
interviews to be audio-recorded and then transcribed (refer to Appendices B and C).
4.4.5.2 Data management.
Source documents provide evidence for the existence of the participant and substantiate the
integrity of the data collected (Lincoln & Guba, 2000). Once transcribed, audio-recordings were
destroyed and all transcripts were de-identified by numerically labelling documents and removing all
identifiable information. In my research, the data are considered partially de-identified due to the
relatively small size of the CF population in Australia. As further measures of confidentiality, consent
forms were kept separate from all data and all transcripts were protected in a secure environment at
The Telethon Kids Institute. All external computer files were password-protected. Only I as the lead
researcher, and named members of the research team had access to confidential records. I ensured
all documents pertaining to this study were made available to the relevant regulatory authorities. I
complied with applicable privacy and security laws for use and disclosure of information related to
the research set forth in this protocol. My research was conducted under the auspices of the AREST-
CF psychosocial research group and within the governance structure and research experience and
expertise of AREST-CF. De-identified results and the written report were made available to all
participating parents. Following the required storage period, as specified by the National Health and
Medical Research Council, all data (including recordings, transcripts and demographic information)
will be erased from the computer and external devices. Refer to Figure 3 for an outline of data
analysis and data management processes.
88 Parenting Children with Cystic Fibrosis
Figure 3. A conceptualisation of data management and data analysis processes
4.4.5.3 Informed consent process.
Informed consent was obtained from parents prior to their interview. This was achieved by
providing them with an information statement about the nature of the research, what was expected
of them as participants, how the results of the research were to be used, what information about
them would be recorded, how the data would be stored and that they could withdraw from the
research at any time without giving a reason. Assurances were also given that personal information
would be kept secure, that any publication would not identify individual participants, and that all
aspects of the Data Protection Act and other relevant legislation would be complied with. To further
ensure confidentiality, parents were informed that their real names would not be used in any part of
89 Parenting Children with Cystic Fibrosis
the analysis process (transcript to report dissemination) and would be substituted with a non-
identifiable number code (Corbin & Strauss, 2008). When I met with parents for their respective
interviews, an information statement was given to them at which time they signed an informed
consent form. Prior to telephone interviews, parents were required to sign and send their consent
form back to me.
4.4.5.4 Consequences of the research.
The nature of qualitative research, and in particular the method of interviewing, lends itself
to openness on behalf of participants whereby personal and/or intimate information may be
disclosed. Expressing information of this nature can potentially arouse mixed emotions about
sharing such information (Kvale & Brinkmann, 2009). Furthermore, it was recognised that the
conversation might be uncomfortable and remind some parents about unpleasant experiences to
which they may have had an adverse reaction. To manage such issues beyond debriefing and
provision of available psychological support at the conclusion of each interview, I ensured that
parents guided the conversation.
4.4.5.5 Role of the researcher.
Conducting research in a morally responsible manner is based on more than ethical
obligations and methodological decisions and acknowledgements (Kvale & Brinkmkann, 2009).
Particularly in qualitative research involving interviews where the researcher is often the main
instrument of data collection and thus obtaining knowledge, the moral integrity of the researcher is
of upmost importance (Creswell, 2007; Kvale & Brinkmann, 2009). Given epistemological and
ontological underpinnings of the research, my interpretations were explicitly informed by reflexivity,
which is introspection and acknowledgement of values, history, interests and potential biases, and
typifies qualitative research (Balbi, 2008). The personal-self and the researcher-self become
inseparable, and acknowledgement of this concept represents honesty and openness to research by
acknowledging that all inquiry is laden with values (Mertens, 2012). Reflexivity emphasises the
importance of self-awareness and ownership of one’s perspective (Patton, 2002). I exercised
reflexivity by attempting to understand how my own experiences and background affect what I
understood (Liamputtong & Ezzy, 2005), to minimise researcher effects on data collection, analysis
and interpretation. Firstly, I systematically reflected on my role during inquiry (both during
interviews and analyses) and continually considered my personal biography and how it potentially
shaped my research. Secondly, by acknowledging potential power differentials, I was able to view
the interview process as a learning opportunity (Reason & Bradbury, 2008). Lastly, I applied the
practice of bracketing during data collection, analysis and interpretation (Braun & Clark, 2006).
Similar to reflexivity, bracketing is a strategy for ensuring that any and all extrinsic influences
are identified and duly acknowledged. In practice, it refers to a process of identifying, acknowledging
90 Parenting Children with Cystic Fibrosis
and suspending preconceived opinions and beliefs about the phenomena under study (Patton,
2002). This allows the researcher to bracket out presuppositions in an effort to confront their data in
pure form (Polit & Beck, 2014). Bracketing was achieved through application of a number of
mechanisms. Firstly, I am not a parent and was consequently able to bracket preconceived notions
about parenting and parenthood. Secondly, I was a novice of the AREST CF collaboration and I
therefore learnt about the surveillance procedures early in the research process, and learnt about
parental experiences as part of data collection, analysis and interpretation. Therefore, by being self-
aware and reflexive in consciousness, and by bracketing preconceived ideas about the phenomenon
under investigation, scientific rigour of interpretation was ensured. Finally, in an attempt to reflect
parents’ stories accurately and sensitively, retrospective rigour strategies were utilised to ensure
validation of interpretation (Creswell, 2007), which was discussed in detail in the previous section of
scientific rigour techniques.
4.4.5.6 Additional ethics considerations.
In the interest of both ethical treatment of participants and duty of care on my behalf as a
researcher, parents were provided with a list of counselling services they could contact if, after the
interview, they felt distress or concerned about anything we discussed during the interview (refer to
Appendix D for Western Australian services and Appendix E for Victorian services). Lastly, it is
important to note that no respondents were denied an interview, that is, all parents from the
eligible sample of 131 families who responded to the research invitation were interviewed.
Outcomes from the interviews are presented in the next chapter. How interviews were conducted is
the topic of the next section.
4.5 Data collection
4.5.1 Interview procedure.
The selection of an appropriate data collection method is primarily dependent on the
research questions (Chwalisz et al., 2008; Kvale & Brinkmann, 2009; Patton, 2002). There are two
key experiences of interest in my research; parents’ experiences and psychological constructions of
parenting in the context of early surveillance for CF lung disease, and how parents’ cope with their
experiences (refer to Figure 4 for an illustration of outline of data collection). To adequately explore
these, a semi-structured interviewing technique, that is, one that provides a set of questions to ask
about the topics but also allows the conversation freedom to vary substantially between participants
was adopted (Fylan, 2005; Wengraf, 2001).
91 Parenting Children with Cystic Fibrosis
Figure 4. Illustration outlining data collection process
To develop rapport with parents (Kvale & Brinkmann, 2009), I first engaged each parent with
some demographic and family questions such as age, number of children, and child care utilisation
(refer to results chapter for demographic data categorised by site and stratified age). This
information also provided a context for the findings emerging from interviews. Whilst all questions
in the interview schedule were addressed with each parents, a flexible approach was taken to the
order of questions, and further issues raised by parents were explored (Playle, 2000).
A total of 14 interviews were conducted with both mother and father present. To achieve
the maximum number of participants in the most convenient way possible for the parents,
interviews with either mother or father present, or both present together were accepted. A
potentially positive consequence or outcome is that discussing thoughts and feelings together may
have helped parents to express themselves more so than if they were interviewed alone. Limitations
of this technique are explained in chapter seven.
The telephone interview became an integral data collection method in this study because it
became apparent in the early stages of recruitment that for some parents, face-to-face interviews
92 Parenting Children with Cystic Fibrosis
were either challenging or inconvenient. Some parents were concerned about infection control
because people with CF should not interact with other people with CF due to the likelihood of cross-
infection, meaning that parents did not want to bring their child to the clinic for an extra visit. For
other parents, home visits were inconvenient due to geographical location of residence or time
and/or work constraints, or because their child may have been experiencing ill health. Whilst
changing the environment of the interview, the content of the telephone based interview continued
to be conversations about parents’ experiences and I was able to probe deeply and seek clarification
from parents regardless.
Parents interviewed via the telephone were willing to discuss their experiences as much as
those parents who were interviewed in person. Therefore, quality and quantity of telephone
interview data was not noticeably different from face-to-face interview data. Furthermore, by using
telephone interviews as an alternative medium in conjunction with face-to-face interviews,
increased recruitment was achieved whilst not compromising the method and philosophy of
constructivism. Had telephone interviews not been offered, 17 parents’ voices would not have been
heard.
I stopped one interview due to apparent emotional distress of a mother expressing a
particular event. She was offered the opportunity to finish the interview and I reminded of her right
to withdraw from the interview and research. However, she was willing to continue with the
interview. At the conclusion of this interview, the mother was reminded of the professional services
available to her and encouraged to contact them if she so felt the need. The parent commented that
she felt relieved about being able to express her emotions about the particular event. Other parents
also described feelings of well-being and relief following the chance to express their thoughts and
emotions about a difficult topic. The opportunity to share their experiences as well as their thoughts
and feelings was mentioned by some parents as providing a type of therapeutic and beneficial
effect, as they felt they had gained insight into their experience and relieved to express and share
their feelings.
At the completion of each interview, parents were thanked for participating, reminded of
my ethical obligations as a researcher and offered the chance to ask questions. To reduce risk of
discomfort or distress as a result of being interviewed, regardless of how I perceived the interview, a
debriefing with parents followed each interview. Parents were debriefed by allowing time to discuss
any concerns they may have about the interview or any other aspect of the research. In the case of
issues arising after I had left, a list of local counselling services was given to each parent they could
make contact with. In the case of any issues arising for parents, I would have organised to follow up
any concerns with each CF clinic coordinator who would then contact relevant support services
93 Parenting Children with Cystic Fibrosis
through their community network connections; however no parents chose to contact either CF clinic
coordinators from each site about the research.
All interviews were between 30 and 90 minutes in duration, depending on how much
information parents were willing to share with me. The primary reason for variation in length of
interviews was how much experience parents had with AREST CF and the hospital environment.
Their amount of experience was related to the age of their child and therefore exposure to early
surveillance of CF. Other potential reasons for variation include amount of time available to be
interviewed, level of distraction, capacity to articulate experience or level of willingness to share
their experience (Morse, 2000). However, provided that data saturation is achieved, variation in
length of interview is not a concern (Francis, et al., 2009).
Once I had finished each interview, I recorded personal impressions and reflections in a
journal. This included notes about responses that generated interesting or relevant information for
further consideration. The journal was a key element of the audit trail important for data analysis
(see the last section in this chapter for further information on appropriate qualitative validation
strategies). The journal was used to support data analysis by assisting with further delineation and
refinement of responses to interview questions, and also by providing further context to the
emergent findings. Please refer to Appendix F for a number of samples from my personal reflections
written in my journal after each interview. To clarify misunderstandings in the data and also to check
for verification of interpretation, follow-up telephone interviews were conducted and several e-
mails were exchanged with seven parents.
4.5.2 Data collection instrument.
To obtain the interpretations of each parent’s personal experiences, interviews were
facilitated by an interview schedule (Smith, 2003). I developed a series of interview questions
designed to answer the research questions that were piloted with the AREST CF parent
representative group, a consumer reference group formed as part of a larger AREST CF psychosocial
research group. The group consisted of five parents whose children had previously been part of
AREST CF but who were no longer under surveillance due to their older age, and one adult with CF.
The pilot was conducted to determine appropriateness and effectiveness of questioning in being
able to elicit relevant information to address the research questions (Kvale & Brinkmann, 2009;
Smith, 2003). A digital recorder was used to record each interview for the purpose of reviewing the
interview narrative. As a result of this process, a number of questions were amended before
commencement of data collection (Porter & Carter, 2000), for example, an initial question, ‘are
there any improvements to the service that could be made?’ was further specified to ‘is there
anything the hospital can do to better support you or your child?’ The interview schedule was then
94 Parenting Children with Cystic Fibrosis
reviewed by a staff member at the School of Psychology and Social Science, Edith Cowan University,
to determine face validity and suitability of questions.
The modified interview schedule (refer to Appendix F) included a set of semi-structured
questions designed to address the research questions that I then used to guide interviews. The semi-
structured interviews explored parents’ experiences associated with their child’s involvement with
early surveillance. Additionally it explored broader experiences associated with their child’s CF;
what coping means for caregivers involved in early surveillance, what coping strategies are
employed for each of the clinical components of early surveillance, and whether coping evolves over
time. Whilst all questions were addressed with each parent, a good deal of flexibility in the order of
questions and exploration of further issues raised by each individual parent occurred (Playle, 2000).
The design of the interview elicited information specifically about parents’ thoughts,
feelings, and perceptions related to the systems and processes, practicalities, and receipt of health
information involved with each annual surveillance assessment, as well as the broader impact of
early surveillance of CF on individual and family life. The interview format was consistent with
Smith’s (2003) suggestion that an interview should begin more generally and become more specific.
In addition, probing questions were used at times when I wanted to further explore issues raised by
parents, for example ‘can you please tell me more about that?’ or ‘can you please give me an
example of that?’
4.6 Exploratory analysis
My research adopted and adapted Braun & Clarke’s (2006) six-step guidelines to thematic
analysis for qualitative psychological research, which reflect Giorgi’s (1985) stages of
phenomenological data analysis outlined earlier in the chapter. These phases are: familiarisation
with data, generating initial codes, searching for themes among codes, reviewing themes, defining
and naming themes, and producing the final report. Whilst not prescriptive (Rapley, 2011), the
primary objective of following Braun and Clarke’s (2006) guidelines was to ensure that an exhaustive
and systematic analysis of qualitative data was conducted. Therefore, it is important that the phases
are recognised and used as a guide rather than a linear or hierarchical approach so as to allow
flexibility to fit the research questions and data (Elliot & Timulak, 2005; Patton, 2002). In practice,
the aim was to approach analysis of raw data in a procedural manner which produced a rich
description of meanings constructed by parents but which did not hinder the ad hoc, unique,
evolving and unanticipated revelations often encountered through qualitative inquiry (Liamputtong,
2009). Accordingly, though there are six main phases in thematic analysis, data collection and
analysis occurred simultaneously and the operations in each phase used as tools rather than
prescriptive steps, thereby retaining the dynamic nature of qualitative research (Corbin & Strauss,
2008). A brief outline of the adopted and adapted guidelines suggested by Braun & Clarke (2006) is
95 Parenting Children with Cystic Fibrosis
presented in Table 3, along with a short description of requirements at each phase and outcomes
from following the process within each phase.
Table 3. Stages of thematic analysis for qualitative psychological research
Stage Process Result
Stage 1 – Familiarisation with data
Read and re-read data, paying specific attention to recurring patterns.
Initial codes and detailed notes.
Stage 2 – Generating initial codes
Document where and how patterns occur by collapsing data into labels to create categories. Data complication is also completed here that involves making inferences about what the codes mean.
Comprehensive codes of how data answers research question.
Stage 3 – Searching for themes
Combine codes into over-arching themes that accurately depict the data and gather all data relevant to each potential theme.
List of candidate themes for further analysis.
Stage 4 – Reviewing themes
Determine how the themes support the data and the overarching theoretical perspective. If the analysis seems incomplete, there is a need to go back and find what is missing.
Coherent recognition of how themes are patterned to tell an accurate story about the data.
Stage 5 – Defining and naming themes
Definition of what each theme is, which aspects of data are being captured, and what is interesting about the themes.
A comprehensive analysis of what the themes contribute to understanding the data.
Stage 6 – Producing the report
Decisions must be made about which themes make meaningful contributions to understanding what is occurring within the data. Member-checking should also be completed here.
A thick description of the results.
Note. Adapted from “Using thematic analysis in psychology,” by V. Braun and V. Clarke, 2006, Qualitative Research in Psychology, 3(2), p. 87.
Phase 1: Familiarity with the data
After interviews were conducted, I transcribed them as a way of becoming familiar with the
data (Braun & Clarke, 2006; Flick, 2006). Listening to audio-recordings during this process allowed
me to immerse myself in the data early on, which provided a valuable way of becoming familiar with
the data (Braun & Clarke, 2006; Elliot & Timulak, 2005). To become familiar with the depth and
breadth of content, all transcripts were read as a whole for deeper immersion in the data (Braun &
Clarke, 2006). Reading transcripts in this way served a number of purposes. Firstly, it contextualised
the data, which gave me an overall sense of parents’ experiences. Secondly, it enabled me to both
conserve the meaning of the data as a whole as well as start to consider structure of meanings
within different interviews and across genders and age groups. Thirdly, it allowed me to begin
96 Parenting Children with Cystic Fibrosis
identifying, delineating and synthesising descriptive accounts which expressed the structure of
meaning attached to the phenomenon under investigation. Lastly, I was able to raise questions
about the continuity and contradictions of accounts generated by accessing multiple information
sources.
Phase 2: Generating initial codes
Coding has been described as a procedure that breaks large volumes of data into smaller
manageable categories (Liamputtong, 2009). At this point, the first interview was read several times
for further familiarisation of the breadth and depth of the content (Braun & Clarke, 2006), and
potentially salient words and phrases were developed into codes to provide meaning to the
descriptive information (Miles & Hubermann, 1994) and then recorded. The data were then
reduced by grouping similar categories and codes, and identifying the most salient themes. As codes
were connected and clustered together, the transcript was checked for accuracy. These themes
were then recorded at points of text that represented it (Liamputtong & Ezzy, 2005; Smith, 2003).
This process was continued with subsequent interviews. Codes from previous cases were utilised to
orient subsequent analyses, respecting convergences and divergences within the data. Constant
comparative analysis was applied to develop and check for accuracy of theme boundaries, which
involved comparing subsequent transcripts to previous ones, using identified codes from the first
transcript to analyse the rest (Janesick, 1994; Strauss & Corbin, 1994).
The entire dataset was worked through systematically as outlined above allowing equal
attention to each transcript. The coding process was conducted with the qualitative analytic
computer software package NVivo 10 whereby codes were created and text collated from each
interview transcript that represented them. With collation of text, much of the surrounding data
were retained so as to ensure that context of the coded data was not lost (Elliot & Timulak, 2005).
This process formed the basis for repeated patterns of themes to form across the dataset (Braun &
Clarke, 2006, Liamputtong, 2009). Sub-themes that did not automatically fit within boundaries of
major developing themes were kept as potential fragments of a major theme that may have
represented a major theme not yet identified (Braun & Clarke, 2006). Storing large sections of text to
a code and retaining sections of text that did not automatically fit into the emergent structure
ensured that inconsistencies and contradictions were not ignored across the dataset, instead
informing the overall analysis (Braun & Clarke, 2006; Grbich, 2007; Kitto, et al., 2008; Liamputtong &
Ezzy, 2005; Mays & Pope, 2000; Morrow, 2005). As identified earlier, it is important in qualitative
research to ensure that uniqueness of people’s experiences is represented beyond generalisations of
these accounts. This is important for facilitation of credibility and authenticity of the findings
because condensations and interpretations of the data become more transparent, meaning
established arguments from the most probable interpretations presented are made clear, therefore
97 Parenting Children with Cystic Fibrosis
allowing the reader to look for alternative interpretations (Grbich, 2007; Kitto, et a., 2008;
Liamputtong & Ezzy, 2005; Mays & Pope, 2000; Morrow, 2005).
Phase 3: Theme development
Following identification of codes within the data, underlying meaning and construction of
experience within codes eventually required consideration on an interpretative level (Elliot &
Timulak, 2005). Underlying threads of meaning and construction from codes are called themes that
aim to describe aspects within the structure of experiences that are neither objects nor things, but
rather address the how or the why of experience (Liamputtong, 2009).
The process of theme development involved exporting data from the NVivo computer
software package so as to manipulate data manually. From this point on, I was able to more
intimately connect with the data to define and order conceptual interpretations in more nuanced
ways than NVivo allows. This was made possible by cutting up sheets of paper with codes and their
descriptions, and organising them across larger sheets of paper. This began development of
categories (refer to Appendix H). Making the process visual in this way made it more dynamic and it
allowed for movement back and forth of compelling statements and ideas across emergent themes.
From this process, I was able to review the level of the coded data extracts, reading all collated data
for each theme together to identity and justify a coherent pattern of thought. Essentially, this
allowed analysis of the already coded data to be compared and considered in combination to form
overarching themes (Braun & Clarke, 2006).
I decided to obtain a detailed thematic description of the entire data set, rather than a more
detailed and nuanced account of one particular theme within the data. This enabled me to develop a
sense of the predominant themes within the data, therefore reflecting an accurate representation of
the entire data set (Braun & Clarke, 2006). Themes were identified using an inductive or bottom-up
approach, which means that themes were identified without a priori development. This involved
bracketing analytic preconceptions (epoche) about the phenomena based on previous literature
(Crotty, 1996).
Phase 4: Reviewing themes
Several thematic mind-maps were created to consider how different codes may combine to
form overarching themes (refer to Appendices I & J). These provided graphical representation of
links and associations between codes and themes (Braun & Clarke, 2006). Once a candidate thematic
map was decided upon, the themes were then reviewed for internal homogeneity and external
heterogeneity; this meant that themes needed to cohere meaningfully whilst still retaining
identifiable distinctions between themes (Patton, 2002). All collated extracts for each theme were
considered for whether they appeared to form a coherent pattern representative of each theme. In
considering relationships between codes, themes and different levels of themes, some codes were
98 Parenting Children with Cystic Fibrosis
found to fit into more than one topic area, which prompted generation of master themes containing
sub-themes. This process further assisted in verification and review of any existing themes (Braun &
Clarke, 2006; Rapley, 2011).
Once I was satisfied that the candidate themes adequately captured the contours of the
coded data, consideration of the validity of individual themes in relation to the entire data set
occurred through accurate reflection of the meanings evident in the data set as a whole. Established
themes and related sub-themes were contrasted with the overall story told across the entire
dataset. Additional coding of data within themes that had been missed in earlier coding stages also
occurred at this stage (Braun & Clarke, 2006). New themes were added and some themes were
dropped as they either did not fit well in the emerging structure, or were not very rich in evidence
within the transcripts.
Reflective writing and member-checking became invaluable during this process. Firstly,
reflective writing pointed to initial arguments to support developed conclusions, and how those
conclusions were reached. The exercise of continuous writing in an ordered format also forced
clarification of ideas (Bazely, 2009). Secondly, member-checking of emergent themes meant that
reconstructions of a reality were confirmed as precise and truthful representations of parents’
multiple realities. This was achieved by conferring with a methodological expert for confirmation of
appropriate them development.
At this point, I was satisfied that the themes accurately represented the essence of the
coded data. It was then that the literature was re-engaged to enhance sensitivity to subtle nuances
arising in the interview data (Corbin & Strauss, 2008). This meant that where themes, associated
sub-themes and concepts already existed in the literature, comparisons of their similarity and
significance to experience could be noted (Braun & Clarke; Corbin & Strauss, 2008). By engaging in
literature at this point, I could practice bracketing, or epoche, of subjectivities to maintain sensitivity
to the interview data (Corbin & Strauss, 2008; Morrow, 2005). The nature of questioning during this
process was how concepts were similar, or different, to those discussed in the literature (Corbin &
Strauss, 2008).
Phase 5: Defining and renaming themes
By this stage of thematic analysis, central themes and their associated sub-themes had been
identified and allocated working titles. However, it is through this phase that nomenclature used in
the final analysis was decided. Clarification of theme and sub-theme names was undertaken and
generation of a description of each theme and sub-theme developed during this phase. This was
achieved by analysing data within each theme, identifying the essence of each theme and
determining which aspects of that data each theme captured (Braun & Clarke, 2006). A detailed
99 Parenting Children with Cystic Fibrosis
analysis was written and consideration given to how each theme fits into the overall understanding
of the data in relation to the research questions (Braun & Clarke, 2006).
The defining and naming phase involved consulting with a number of professionals from the
multi-disciplinary CF care team at PMH. This served to triangulate data interpretation thereby
ensuring credibility of identified themes and sub-themes (Vicary & Bishop, 2005). Compelling
statements in the form of vivid quotes were carefully selected and intentionally collated to convey
the essence of a theme and its associated sub-themes presented in the next section (Braun & Clarke,
2006). The consistency and truthfulness in this process resided with my ability to link abstracted
themes and sub-themes drawn for the dataset, bringing alive for the reader an in-depth sense of
how parents whose children have CF construct meaning of their child’s early disease surveillance.
Phase 6: Producing a report
The goal of thematic inquiry is to illuminate the essential, structural qualities of a particular
experience (Becker, 1992). This was achieved through the resultant set of themes that were derived
from the parents’ interviews, the research questions, theoretical frameworks and philosophical
assumptions. A qualitative approach to analysis consisted of identifying an experience to explore,
acknowledging and positioning my own assumptions and biases, collecting data from individuals
who have experienced the phenomenon, analysing collected information by reducing it into relevant
assertions and quotes, and developing a textural description (i.e., what parents experienced), a
structural description (i.e., which contexts or situations have typically influenced or contributed to
parents’ experiences of their child’s early disease surveillance) and also a combination of the two to
convey an overall essence of the experience (Creswell, 2007; Moustakas, 1994). Willig (2001)
suggested a format of structured themes and sub-themes with their descriptions and quotations to
illustrate each theme.
The next chapter details the outcomes of this process. Whereas the previous chapter
explained what scientific rigour is, its importance in qualitative research and the strategies used to
achieve it, the next section details the activities I undertook to ensure rigorous techniques were
adhered to.
4.7 Scientific rigour
The discussion will now turn to how rigour was achieved according to different stages of the
research; data generation, coding and analysis, and presentation of findings, beginning with a short
discussion of strategies that occurred throughout the entire study (Lincoln & Guba, 1985).
4.7.1 Throughout the research process.
The primary strategies applied continuously to my research were that of reflexivity (Patton,
2002) and bracketing (Polit & Beck, 2014): reflexivity during research design, data collection, analysis
and interpretation; and bracketing during data collection, analysis and interpretation. Applying and
100 Parenting Children with Cystic Fibrosis
maintaining reflexivity and bracketing ensured extraneous influences and preconceived notions
were minimised in the research process.
Throughout analyses, an audit trail (Charmaz, 2000) was created to enhance scientific rigour
by ensuring interpretation followed a logical and conclusive path. An audit trail is documentary
evidence for neutral experts or peer reviewers to review and verify the path the researcher followed
from raw textual data to results. It is used to establish the rigour of a study by providing the details
of data analysis and some of the decisions that led to the findings (Wolf, 2003). According to Rodgers
and Cowles (1993), data for an audit trail can be classified into six categories: raw data, data
reduction and analysis products, data reconstruction and synthesis products, process notes,
materials relating to intentions and dispositions, and instrument development information. The
audit trail then becomes part of the confirmability and dependability process.
4.7.2 Data generation.
At the point of data collection, aspects of ensuring rigour of qualitative research include
representativeness of sample, sampling technique, and researcher reflexivity (Kitto et al., 2008;
Mays & Pope, 2000; Morrow, 2005). I endeavoured to ensure rigour by explicitly describing the way
my research was conducted including detailing methods with regard to accessing and recruiting
participants, and developing rapport and trust with them. How ethical considerations were
addressed also ensured rigourous research methods were employed (Kitto, et al., 2008; Liamputtong
& Ezzy, 2005).
A critical moment in the qualitative research process is engagement with participants.
Therefore, prolonged engagement is the investment in sufficient time to achieve the purposes of the
research. I addressed this requirement by spending time learning about parents’ experiences
through the interview process and was directed by data saturation. Consequently, there was a point
at which I was able to establish that the phenomena under investigation had been adequately
explored, and was therefore confident that the data were representative of the phenomena.
Prolonged engagement with parents was also achieved in Western Australia by spending time in the
CF clinic prior to the interviews and introducing myself and the research to parents. Establishing
rapport and trust in this way ensures credibility and authenticity. The conceivable reliability and
quality of data collected and intimate familiarity with the participants and their worlds (Blumer,
1969) is the process through which the researcher can construct reality (Denzin & Lincoln, 2005).
An additional process for ensuring adherence to rigourous research methods during data
collection involves creating personal reflections after completion of each interview. Field notes are a
form of reflective writing (Charmaz & McMullen, 2011), which can be later examined as part of
analysis, both as a reflection on the interview as well as part of reflexivity and epoche. By ensuring I
reflected upon interviews, as a process and as an experience, credibility and dependability of
101 Parenting Children with Cystic Fibrosis
interpretation is strengthened because of additional and extraneous information to the interview
that can verify its accuracy. Furthermore, when field notes are considered as part of data analysis,
transferability of interpretation is achieved because other researchers can determine usefulness of
findings to a population by examining how the researcher reflected on interviews and determining
their effects on analysis. Therefore, possessing additional information beyond the findings
themselves allows independent researchers to determine for themselves the accuracy in
extrapolation of findings to other populations or contexts.
A number of other techniques described in the sampling and procedure section of this
chapter also refer to strategies of rigourous research methods. Firstly, audiotaping of interviews and
verbatim transcription of interview data ensure credibility and authenticity of data because of the
strict adherence to accuracy in recording data. Secondly, data saturation means that credibility can
be assigned to findings within that population because of the determination that no new data was
being generated, therefore ensuring credibility in the findings. On a final note, previous qualitative
experience ensures rigour during the interview process, specifically credibility and authenticity
because the quality of information obtained is highly dependent on the interviewer (Patton, 2002). I
was able to draw on my previous qualitative research experience, which gave me confidence to
conduct interviews and ask questions, both as prompts and clarifiers.
4.7.3 Data coding and analysis.
To further ensure rigour of the research, how findings were generated was made explicit,
affirming consistency across time and analysis techniques, making the process as repeatable as
possible (Morrow, 2005; Patton, 2002). This was primarily achieved by tracking the research process
from design to interpretation through use of a reflective journal detailing chronology of research
activities, processes and influences (Flick, 2006; Morrow, 2005). The journal was used at this stage of
the research as a critical source of interpretative understanding as concepts were dissected and
explored (Bazely, 2009), which ensured credibility of interpretation because of the reflexive nature
of the process. Confirmability of interpretation is also inherent in use of a self-reflective journal
because of its ability to provide contextual decisions to generation of interpretation. Essentially,
reflexivity refers to how data are co-constructed during collection through the influence of both
researcher and participant, and offers both a methodological and interpretational log of research
decisions (Corbin & Strauss, 2008). I was therefore able to maintain a level of sensitivity to ways in
which I, acting as an instrument of data collection, shaped the collected data (May & Perry, 2011;
Mays & Pope, 2000). Engaging in this reflexive process allowed acknowledgement that research is
never completely objective, and that quality research should result in outcomes that represent as
closely as possibly the context being researched as opposed to the values and biases of the
researcher (May & Perry, 2011; Morrow, 2005)
102 Parenting Children with Cystic Fibrosis
Assurance that research has presented adequate data and findings is largely dependent on
strategies employed during data analysis including verification steps. A number of verification steps
were included in the analysis process; disconfirmation and discrepancy in cases, constant immersion
in data, member-checking, inter-coder checking, peer debriefing, and coherent theme development.
Each of these verification strategies will be briefly described.
By discovering and examining disconfirmation and discrepancy in cases, also known as
negative case analysis (Patton, 2002), I was able to make repeated comparisons and revise key
assertions and categories until they accurately reflected the experiences of parents (Morrow, 2005).
In qualitative research, negative cases cannot be ignored because they can provide hints that might
explain what is happening for the larger sample (Bazely, 2009). Development and refinement of
hypotheses occurred here because by looking at all cases, existence of ideas about the data are
being tested, and refined by adjusting ideas. A conscious effort to search for disconfirmation also
helped to combat any tendencies on my behalf to seek confirmation of preliminary findings and
categories (Kitto, et al., 2008; Liamputtong & Ezzy, 2005; Mays & Pope, 2000; Morrow, 2005). This
process was also aided through constant immersion in the data from; data collection, transcription
of interviews, and continued and repeated readings of the transcripts, listening to the audio
recordings, and reviewing notes and other related data (Flick, 2006).
Member-checking became important at this stage of the research because, as Lincoln and
Guba (1985) highlight, this is the most critical technique for establishing credibility, which refers to
determining accuracy of findings and interpretations by taking specific interpreted descriptions back
to the participants for verification and clarification. Parents were consulted to ensure that
reconstructions of a reality were precise and truthful representations of their multiple realities. This
was achieved by conducting in-depth follow-up telephone interviews with five parents for validation
of interpretation, as well as sending a brief overview of findings in the form of a newsletter to all
parents providing an opportunity to respond. At this stage of the research, members from the multi-
disciplinary CF care team at PMH were consulted for relevancy of findings to clinical practice and
accuracy of findings based on their anecdotal experiences with families. From engagement with the
multi-disciplinary care team and further engagement with parents, truthfulness of findings and
uniqueness of concepts was substantiated; addressing requirements of confirmability.
Employing inter-coder checking became an important strategy in verifying data analysis.
Inter-coder checking is a process of determining congruence between two or more independent
people about the data’s accuracy, relevance or meaning by employing an external auditor to code
and interpret a portion of the data(Lincoln & Guba, 2000). To assess inter-coder reliability, a fellow
research colleague was asked to code 10 interviews. Comparisons of coding techniques and
103 Parenting Children with Cystic Fibrosis
categories determined accuracy of coding and corroboration of interpretation, addressing
requirements of credibility and confirmability.
Interaction with an independent person for peer debriefing during data analysis was another
verification strategy. Continual interaction with a peer throughout the research process, both about
the research as well as about my role within the research, gave multiple perspectives of the data,
assisting me to develop and confirm ideas and interpretations. This is where my biases were
questioned, meanings explored and interpretations clarified, consequently ensuring credibility in the
process and therefore also in the findings. Debriefing in this way also propels discussion about initial
opportunities to develop working hypotheses. My choices and subsequent decisions were subject to
rational and logical explanation of their position and suppositions (Lincoln & Guba, 1985). An
example of the progress made within debriefing sessions was how my interpretation of parents’
knowledge of their child’s CF disease progression positively influenced their everyday life worlds
beyond the immediate trauma of gaining this insight.
Lastly, verifying the data through theme development included grounding categories and
themes in examples and organising categories to provide coherent understanding of how they fitted
together so as to provide a data-based narrative an underlying structure to organise the
phenomenon for the reader (Elliot & Timulak, 2005; Morrow, 2005). Moreover, the results should
exemplify symmetry of interpretation and quotation by striking a balance between the researcher’s
interpretations and supporting evidence by way of quotations from participants. Providing actual
words of parents’ accounts therefore became essential to informing the reader that the
interpretations made were grounded in the lived experiences of the participants. Furthermore,
providing a clear account of processes from research design and data collection to analysis through
use of diagrams to aid understandings, explicitly outlining procedural information, and engaging in a
number of peer auditing procedures, increased dependability of the research (Flick, 2006; Guba &
Lincoln, 1994; Mays & Pope, 2000: Seale, 1999).
4.7.4 Presentation of findings.
By studying a group of people intensively so as to understand their life-world of the
phenomena under investigation, there will be findings that are unique to that sample
(isomormorphism), but detail can be extrapolated to other similar populations and/or within similar
contexts, resulting also in transferability of the findings (Patton, 2002). Provision of rich, thick
descriptions as adequate evidence of any assertions, such as provision of appropriate quotes to
support themes (Braun & Clark, 2006) and by providing working hypotheses together with a
description of time and context of when such suppositions were found to be valid (Lincoln & Guba,
1985), achieves transferability because it gives the reader the opportunity to determine the
transference of findings to similar individuals or contexts. Therefore, by providing extensive and
104 Parenting Children with Cystic Fibrosis
diverse sources of interpretation, the function of transferability occurs when the reader has
adequate information to determine it. When the research report is sufficiently detailed for the
reader, they can judge whether findings are applicable to similar settings and how transferable,
relevant, and useful the findings are to the particular context and phenomena under investigation
(Kitto, et al., 2008; Mays & Pope, 2000). By employing strategies discussed in this section, I have
presented sufficient information so the research could be considered relevant, transferable, and
useful to the research and clinical context of early surveillance and CF care more generally.
Accordingly, quality or validity in qualitative research is, to a certain extent, paradigm-bound
with standards of trustworthiness that emerged from paradigms such as those in quantitative
research (Kitto, et al., 2008; Lincoln & Guba, 2000; Mays & Pope, 2000; Morrow, 2005; Seale, 1999).
However, particular qualities are also indispensable regardless of the research paradigm such as
reflexivity, and issues related to adequacy of data and to interpretation and presentation of data
(Alvesson &Sköldberg, 2009; May & Perry, 2011; Morrow, 2005). Accordingly, as in quantitative
research, rigour and quality in qualitative research achieved through systematic, self-conscious
research design, and data collection, interpretation and presentation was required (Flick, 2006;
Mays & Pope, 2000). By using those strategies and techniques outlined above, I achieved a
transparent and thorough account of the research process and associated findings.
4.8 Summary
The methods chapter has outlined qualitative strategies and techniques I used to achieve my
research aims and address my research questions. My sample, and how I recruited them, was
detailed, along with ethics considerations I made throughout the research process to ensure quality
of data. More importantly, ethics considerations ensured ethical conduct of the research and
appropriate consideration for my participants. Lastly, data collection procedures and data analysis
techniques were outlined. The format of presenting structured themes and sub-themes is utilised in
the next chapter where a composite description of parents’ experiences, along with a discussion of
findings and interpretations are presented. Below is an overview of the theoretical and procedural
frameworks guiding the research processes that have been explained throughout the chapter.
Figure 5. Overview of theoretical and procedural frameworks guiding the research
105 Parenting Children with Cystic Fibrosis
CHAPTER 5
Findings: The lived experience of parenting children with CF in the context of early surveillance
Chapter 5 presents the findings and interpretations of this research. The aims were to explore and interpret parents’ experiences and psychological constructions of parenting. Further aims were to explore how parents cope and what meanings were attributed to their experiences. Salient findings from analysis of the data are the commonalities in experiences and constructed thoughts about parenting children who undergo early surveillance for CF lung disease. Accordingly, the chapter presents the temporal experience from diagnosis through to day-to-day management of CF within context of early surveillance, through the first 7 years of life. The chapter begins by outlining the major themes and sub-themes identified from the data analysis process. Each theme within this chapter commences with an outline and summary of the identified major theme and continues with a discussion of related sub-themes and presentation of excerpts from participants’ responses to demonstrate the findings and interpretations. Throughout each section, current research and literature relevant to the identified themes and sub-themes are integrated into the discussion. Interpretation of findings in light of applied literature consolidates their place within the lived experience of parenting children with CF in the context of early surveillance.
106 Parenting Children with Cystic Fibrosis
We must be willing to let go of the life we have planned, so as to have the
life that is waiting for us…Joseph Campbell
5.1 Identifying Themes
A primary aim of my research was to explore how parents of children with CF residing in
Perth, (Western Australia), and Melbourne (Victoria) experience and construct parenting within
context of early surveillance for CF lung disease. Further aims were to explore how parents cope and
what meanings were attributed to their experiences. Lastly, my research aimed to review all
participant groups’ experiences collectively to identify any unique commonalities and/or differences
in experiences and constructed thought. Through the process of thematic analysis (as outlined in
chapter 3: Methods), a substantial finding from analysis of the data was the commonality in
experiences and constructed thoughts about parenting a child with CF within the context of early
surveillance across the age range of children and across sites. Before I introduce you to parents in
my research, it was evident that their overall experience of CF was inextricably tied to their early
surveillance experience (which I will explain in due course). Therefore, to understand parents’
experience of early surveillance was to also account for parents’ experiences of CF. Therefore;
whilst I analytically separated these two elements of consciousness, I did not attempt to
experientially separate them because they are part of the same thing.
Within the process of analysis, it became apparent that themes naturally cohered in such a
way that parents’ experience preceded from diagnosis of their child’s CF and their introduction to
early surveillance through to their everyday regular and routine experience of parenting in the
context of early surveillance. This was followed by distinct experiences and outcomes of early
surveillance. Accordingly, presentation of themes will follow this emergent structure.
Five major themes with related sub-themes represent the emergent structure: redefined
expectations and reimagined identity, redefined reality (consisting of sub-themes - collapsed and
redefined expectations and new normal); understanding the unknown and understanding
uncertainty (consisting of sub-themes – understanding CF and understanding early surveillance for
CF lung disease); good days and bad days; fluctuation between positive and negative outlook on life
(consisting of sub-themes – bad days, good days and fluctuation between good days and bad days);
early surveillance is a significant event (consisting of sub-themes - indicator of treatment efforts and
examination of parenting competence, parental outcomes directly from early surveillance and fear
from diagnosis exacerbated at each annual review can result in anxiety); and early surveillance is in
the best interest of my child (consisting of sub-themes - perceived clinical benefit to child health and
cognitive dissonance results in anxiety and/or ambivalence). Refer to Table 4 below for an overview
of themes and sub-themes. Consistent with the first three research questions, identified themes
represent salient factors contributing to parents’ experiences and constructions of parenting a child
107 Parenting Children with Cystic Fibrosis
with CF in context of early surveillance, as well as their attributions of meaning to early surveillance.
The fourth research question is addressed throughout themes where coping with diagnosis,
treatment and early surveillance is considered. Each of these themes and their related sub-themes is
discussed separately in the next section.
Table 4. Major themes and related sub-themes
Major theme Related sub-themes
Redefined expectations and reimagined identity; redefined reality
Collapsed and redefined expectations
New normal
Understanding the unknown and understanding uncertainty
Understanding CF
Understanding early surveillance for CF lung disease
Good days and bad days; fluctuation between positive and negative outlook on life
Bad days
Good days
Fluctuation between good days and bad days
Early surveillance is a significant event Indicator of treatment efforts and examination of parenting competence
Parental outcomes directly from early surveillance
Fear from diagnosis exacerbated at each annual review can result in anxiety
Early surveillance is in the best interest of my child
Perceived clinical benefit to child health
Cognitive dissonance results in anxiety and/or ambivalence
5.2 Interpretation of phenomena – what is the ‘lived experience’ of parenting children with CF
within the context of early surveillance for lung disease?
This chapter is a description and interpretation of the lived experience of parenting a child
diagnosed with CF within the context of early surveillance for CF lung disease. Within this chapter, I
identify, describe and explain why discovered phenomena are present for parents. Where relevant, I
will explain how these phenomena function, for example, how situations encountered might affect
parental characteristics and outcomes. The current research landscape investigating psychological
outcomes for parents in paediatric chronic disease is such that resiliency is a key beneficial concept
for parents and their families due to recent focus on resistance, rather than risk factors. Therefore, I
highlight the contribution of early surveillance to parenting a child with CF by identifying risk and
108 Parenting Children with Cystic Fibrosis
resistance factors that may contribute to adaptation. A process to adaptation is to build and
maintain resilient parents and families for future burden of CF, which is continued into the next
chapter. The first theme begins with a description of parenting a child diagnosed with CF.
5.3 Redefined expectations and reimagined identity; redefined reality
Parents whose child is diagnosed with CF, typically within the first 3 months of life, confront
the reality of suffering and the premature death of their child (e.g., Carpenter & Narsavage, 2004;
Priddis, et al., 2010). They can experience a multitude of negative reactions and responses including,
but not limited to shock, disbelief, anxiety, guilt, fear, and depression (De Monestrol, et al., 2011;
Fallowfield & Jenkins, 2004; Glasscoe, et al., 2007; Moola, 2012; Mullins, et al., 1991; Quittner, et al.,
1992). Most studies have concentrated on maternal reactions to diagnosis with less consideration
given to fathers. The starting point for my analysis is that new parents - both mothers and fathers -
operate with expectations about what being a parent means, of what having a family entails. These
expectations hold one’s hopes, aspirations and fears. Once parents are informed that their child, in
whom so many of their expectations are invested, suffers from a progressively fatal condition, home
treatment management and clinical care regimens are swiftly explained. The initially
incomprehensible diagnosis and overwhelming amount of information about treatment and care
affects parents’ expectations, their optimism, their hopes for the future and their perceptions of
themselves as parents in such a way that causes collapse and redefinition of their expectations. This
section relays these accounts through identification of two sub-themes namely; collapsed and
redefined expectations and new normal.
5.3.1 Collapsed and redefined expectations.
Diagnosis of CF in a newborn literally dissolves initial child, parenthood and family
expectations (Bluebond-Langner, 1996; Carpenter & Narsavage, 2004; Jessup & Parkinson, 2010;
Priddis, et al., 2010). Upon diagnosis, emotional despair and heartache for the child, dismantling of
parent identity and collapse of family expectations ensue. Parents’ expectations for parenthood and
family life disintegrate with the diagnosis for their infant, which means that parents were left with
no expectations for their child and their family.
To illustrate, a Western Australian mother of a child aged 4 years reflected on her child’s
diagnosis when she stated ‘you just don’t see that life can have any level of normality when you’re
first diagnosed. You’re worried about all of these things you’re being told you have to do, you don’t
know how you’re going to manage it all. You don’t know what any of this means for your family or
your child’s future.’ The following passage from another Western Australian mother whose child was
5 years old further reflected initial despair experienced by parents about loss of expectations for her
child and family, ‘you just see a little baby who you’re being told is sick, but you don’t really know
how sick, so you automatically think the worst…I think when kids are first diagnosed, it robs you a
109 Parenting Children with Cystic Fibrosis
little bit because it really is a huge shock which shattered our lives and what we expected for our
future.’
Therefore, collapse of expectations about parenthood and family life is a cognitive process
that threatens parents’ expectations and reality irrevocably. The threat functions to diminish
parents’ ability to follow their expected family life path and to believe in what they knew about
parenthood. Jessup and Parkinson (2010) reported a similar process when they suggested that the
future of the family was initially cancelled at diagnosis. Parents experience loss of their previously
taken-for-granted world. They fear the potential loss of their child and fear failing their child through
their inability to conduct the required home treatment regimen thrust upon them at diagnosis
(Cohen, 1993; Jessup & Parkinson, 2010).
Parents are required to learn about the time-consuming and often difficult home treatment
regimen soon after the baby’s diagnosis. When parents begin to experience the intensity of home
treatment and the time required to complete it, their expectations about what family life will be like
are further altered, particularly what their parental role will entail (that is, they are adopting a
medical role) (Glasscoe & Smith, 2011; Tong, Lowe, Sainsbury & Craig, 2010) and what their child’s
life will be like with these daily demands. A Western Australian mother of a 2 year old child spoke
about her altered parental role, ‘you know the parents have to become experts on how to deal with it
and how to manage it, and that means that you have to develop quite a detailed understanding of
the lungs and the disease and stuff. So you need training basically.’ Envisaging what a child’s life may
be like was expressed by a Western Australian father of a 4 year old child who stated ‘I remember
when there was about a 3-month period when you (mother) were trying to teach XXA pep
(physiotherapy) and she screamed every time. I remember asking myself ‘what kind-of life is our child
going to have?’ After collapse of expectations surrounding diagnosis of CF, introduction of intensive
home treatment management is likely to contribute to reconceptualisation of expectations of what
family life will be like.
Diagnosis of CF in an infant changes the ideality of parenthood and family life. Using more
conventional terminology from the CF clinical nurse coordinator at PMH (E. Balding, personal
communication, February 27, 2014), “a CF diagnosis moves the goal posts of life for our families.”
This refers to the notion that parents’ preconceived realities and expectations are shifted by
knowledge of their child’s condition because everything that was known and expected about
parenthood and family becomes threatened (Jessup & Parkinson, 2010). The threat is extensive; it
includes their baby (and their future), their ideals, their current understanding of parenthood; life as
they know it and expected it to be. The known and expected becomes threatened because parents’
expectations of the traditional (normal) life trajectory and what they expected of parenthood and
family life are taken away.
110 Parenting Children with Cystic Fibrosis
Qualitative accounts of parenting children with leukaemia (Kars, et al., 2008), congenital
heart disease (Rempel, Ravindran, Rogers & Magill-Evans, 2013), Duchenne muscular dystrophy and
Rett syndrome (Gravelle, 1997) reported similar psychological experiences. Accepting and adjusting
to the inconceivable reality of their child’s condition was the first process for parents. This involved
two sub-processes; realising the precariousness of survival and adjusting expectations. Constantly
living with pressure and stress of the unknown in terms of their child’s disease trajectory of longevity
of life was another process parents from these studies experienced.
Part of expectations about parenthood includes the substance, and meaning, of becoming a
parent. Moreover, perceptions of both parenting practices (i.e., responsibilities) and parenting
functions (i.e., roles) are inextricably tied to how people perceive themselves as parents (i.e., parent
identity). Parental identity refers to how an individual views him or herself in relation to their child
(Maurer, Pleck & Rane, 2001). Therefore, parents’ beliefs and values about their role in their child’s
life will contribute to how they view themselves in relation to the child. Damage to parent identity
due to paediatric chronic illness is a commonly cited outcome in other chronic conditions (Bowes, et
al., 2009; Cashin, et al., 2008; Marshall, Carter & Rose, 2009; Patrick-Ott, 2011; Waite-Jones &
Madill, 2008), but has been insufficiently investigated in CF. Some qualitative studies briefly
mentioned negative outcomes of CF on parent identity but failed to uncover any specific pathways
(Hodkinson & Lester, 2002; Liossi & Evans, 2005). With support from the literature, I have
interpreted two reasons why parent identity may be damaged by a CF diagnosis.
Firstly, reports in the literature describe how parents can feel guilty about their child’s
condition, likely because of the genetic nature of CF (Hodgkinson & Lester, 2002; Marvin & Pianta,
1996; Priddis, et al., 2010; Quitter, et al., 1992), meaning that parents felt as though they were
responsible for giving their child CF (Havermans, et al., 2015). By accepting responsibility of loss due
to chronic illness (in this case, the child’s loss), guilt can ensue (Carpenter & Narsavage, 2004;
Stephenson & Murphy, 1986). Secondly, parents must come to terms with the forced reality of
parenting a chronically ill child, irrevocably altering what parents envisaged parenthood would be
like (Glasscoe & Smith, 2011; Patrick-Ott, 2011; Tong, et al., 2010). Contributing to loss of ideals
about parenting is the requirement on parents’ behalf to complete intensive home treatment,
further altering ideals about the substance and function of parenting.
The latter point is particularly about the substance and function of the role in relation to
parenting a chronically ill child. That is, parents are adopting a medical role that requires them to
conduct medical treatment and monitor child health. It appeared that, due to demand of home
treatment, care-giving formed a significant part of parenting a child beyond usual parenting tasks,
which has been shown in both paediatric CF (Afonso, Gomes & Mitre, 2015; Liossi & Evans, 2005;
Slatter, Francis, Smith & Bush, 2004) and other chronic illness literature (Bjork, Wiebe & Hallstrom,
111 Parenting Children with Cystic Fibrosis
2009; Cashin, etal., 2008; Heaton, Noyes & Sloper, 2005; Kirk, Glendinning & Callery, 2005;
Pelenstov, Fielder & Esterman, 2015; Tong, et al., 2010). The caring role of chronically ill children
often dominates the parenting role because of the need to provide ongoing care for the child (Kirk,
et al., 2005). Therefore, domination of the carer role over the parent role following diagnosis may
result in a loss of the traditional parenting role and damage to self-schemata about perceptions of
parenting until such time that parents can redefine - reimagine - their identity.
O’Connor’s and Barrera’s (2014) model of identity disintegration and reintegration in
paediatric chronic illness facilitates speculation about potential mechanisms through which parent
identity could be damaged and re-imagined. They conceptualised negative perceptions of
parenthood and the parental role and identity as identity disintegration, and the process of identity
reintegration as a positive reframing of parenthood and the parent role and identity. This framework
provides a contextual layer to psychological theory about self-schemata (Piaget, 1952), and their role
in identity, because it outlines what happens when parents assimilate information of CF into their
parental role following their child’s diagnosis.
How parent identity is repaired for parents in my research is supported by this theory and
demonstrated in Slotter’s and Gardner’s (2014) study, where evidentiary support for the threatened
self-concept generates a reimagined self-concept. Together, O’Connor’s and Barrera’s (2014) theory
and the qualitative findings of Slotter and Gardner highlight that encoded information supporting a
threatened self-concept aids in positively reframing parent role and identity. As parenting a child
with CF can result in conflict between roles of carer and parent (Glasscoe & Smith, 2011),
understanding mechanisms for adverse parental outcomes that may be amenable to intervention is
essential to ensure that parents are fully supported.
To summarise, parents must navigate their way to understanding and subsequently
accepting their child’s diagnosis to preserve their own psychological well-being, and to be present for
their child. Negotiation towards understanding and acceptance involves a process of resetting,
rebuilding and redefining parenting and family expectations. A Western Australian mother of a 2 year
old child exemplified this process when she stated ‘when you are first told, when I was first told you
kind-of just go "oh (deflates)," and you're whole plan collapses, and then I think you just start the plan
again in some kind-of re-moulded form.’ This is achieved through psychological absorption and
eventually acceptance of the diagnosis, though it is conceivable that parents come to accept their
child’s diagnosis over various time frames and with varying degrees of effectiveness (Priddis, et al.,
2010).
New normal is the classification of the experiential and practical outcome of redefined child,
parent and family expectations. It refers to the concept of parents’ acceptance and embracement of
a different life to what was expected before the intrusion of CF. New normal is the result of
112 Parenting Children with Cystic Fibrosis
knowledge acquisition and experience accumulation, that is, expectations are redefined based on
understanding and experience, and then parents assess the truthfulness and achievability of those
redefined expectations through further understanding and experience. New normal is the final
outcome of redefined expectations.
5.3.2 New normal.
Through the experience of moving from collapsed to redefined expectations, parents
develop a new sense of normal life, with CF and its management as additional components.
Reconceptualising what is perceived as normal is a common theme in the chronic illness literature,
as demonstrated by a metasynthesis (Nelson, 2002) of qualitative studies about mothering a child
with a disability or chronic condition. Reports have demonstrated that parents of children with CF
strive to maintain a degree of normality within the family domain (Liossi & Evans, 2005, Moola,
2012). Admi (1996) posited a model for living with CF that placed maintenance of a normal life at its
center. Carpenter and Narsavage (2004) described how parents return to normality after the
devastation of a CF diagnosis. Bluebond-Langner (1996) reported that redefinition of normal begins
in the year following a CF diagnosis, and Glasscoe and Smith (2011) published a case study that
detailed how a mother’s awareness of CF and the treatment regimen became part of their everyday
lives for her and her child.
The conceptual structure of what I have termed new normal is relevant for any family or
person who is required to live with a permanent unexpected change to their everyday life world; to
the essence of their existence. It refers to a new way of living that must be negotiated and
eventually accepted as part of their redefined way of living. Therefore, living with a permanent
unexpected change refers to going beyond the change simply existing in one’s life, but encompasses
its successful management and acceptance as part of one’s life.
Jessup and Parkinson (2010) likened this event to parents “needing to find their bearings
and reconstruct their lives in the context of a new, unanticipated scenario (pp. 355).” To that end,
the conceptual basis of new normal for parents in my research, in the context of CF diagnosis, refers
specifically to how those processes of negotiation and establishment of a reimagined sense of
identity (including roles and responsibilities) and redefined expectations reach a conclusion. For
example, a Western Australian mother of a 3 year old child reflected on her transition from
devastation of diagnosis to the normality of the present:
it’s not all bad. I think to begin with it is, but then it gets so much easier from those first
days where I was filled with negative thoughts, to one year on and then 2 years on, it
gets easier… it gets easy and there is a lot of normal because you just don’t see it when
you’re first diagnosed. So just to know that all the things that you’re really scared of
113 Parenting Children with Cystic Fibrosis
become a part of your day-to-day life that you don’t even notice it. You manage, you just
manage.
Similarly defined in the chronic disease literature, normalisation for parents is described as
both a cognitive process of defining their own lives and a set of behavioural strategies for managing
their child’s condition (Knafl, et al., 2010; Rehm & Franck, 2000; Robinson, 1993). Furthermore,
normalisation has been referred to in the CF literature as integrating CF treatment into, yet
maintaining, family routines (Pfeffer, et al., 2003) including cognitions and actions that portray
treatment as a necessary routine within that new sense of family life (Glasccoe & Smith, 2011;
Grosssoehme, et al., 2014; Hodgkinson & Lester, 2002; Jessup & Parkinson, 2010). A landmark study
focussing on psychological outcomes of CF in the family environment by Bluebond-Langner (1996)
identified strategies used to manage the early stages of diagnosis that offered avenues for achieving
a sense of normality including routinisation of treatment into daily life, redefinition of what is
perceived as normal and reconceptualisation of the future.
Two processes simultaneously occur that function as mechanisms for negotiation of parent
and family expectations. Firstly, parents are gaining knowledge about CF, the condition, its
management, their role as parents of a child with CF; their individual and collective futures.
Secondly, parents are beginning to have experiences of each of these aspects of their child,
providing evidence of their ability to meet these new expectations and requirements. Whilst
absorption of this type of knowledge, and early experiences of CF, can be initially overwhelming and
devastating, it begins to give parents a framework upon which to build understanding of their child’s
condition. Very few parents have such a framework at diagnosis due to their lack of awareness that
the condition is possible within their family, as well as the broader lack of public awareness of CF at
the societal level.
Keeping in mind the supposition that early surveillance is inherently linked with CF, it too
becomes part of the new normal for families in my research. The introduction of early surveillance
for CF lung disease soon after diagnosis, along with parents’ perceived hasty requirement to notify
their involvement, results in the amalgamation of these two entities as part of the same experience.
Whilst the disease of CF can be distinguished from the experiential encounter of early surveillance,
their distinction in terms of thinking of one without the other is not possible. This is the inextricable
link between the two. Therefore, accepting and embracing life with CF for parents in my research
included accepting and embracing early surveillance as part of their new normal.
Since early surveillance is introduced so close to diagnosis, parents know no other reality of
CF and so have no alternative way of thinking for comparison, and therefore seem to accept what is
presented to them as a routine part of CF (Glasscoe & Smith, 2011). A Victorian father of a child aged
12 months discussed how he viewed early surveillance in relation to his child’s CF; note the tense in
114 Parenting Children with Cystic Fibrosis
the quote moves from the past to the present and therefore seems to define the status quo now,
‘well it’s part and parcel really isn’t it? For us, we’ve known CF with the program, never without it.
We’ve never known any different so it’s not like we can compare to not being part of the program or
whatever, know what I mean. This is our experience of CF and we want to know the information we
get from being part of it (early surveillance). Actually, I can’t really imagine not getting this info
about my kid’s health, it’s valuable because it can help her have a better life.’
In early stages of diagnosis and establishment of the new normal, parents know what early
surveillance entails pragmatically and what is expected of them and their child. However, at this
point there is a lack of personal experience. In these early stages when parents are negotiating and
accepting a redefined sense of family life, there is little consideration of how they will manage their
personal early surveillance experience. Until such time that parents can have their first encounter
with early surveillance, they cannot start to understand how to manage the personal experience.
This understanding from experience will be discussed comprehensively in the next theme, because
understanding from experience emerged as an interpretation of the overall parental experience of
early-life CF management.
To finish outlining parents’ new sense of normal, a brief reiteration is warranted of the
mechanisms through which parents come to their redefined realities. A redefined sense of personal
and family life emerges through understanding: an understanding that occurs over time with
knowledge and experience. Understanding comes from gaining knowledge about CF and early
surveillance. Accumulation of both knowledge and experience are the essence of accepting and
embracing new normal. Therefore, a sense of new normal is the outcome of the transition from
devastation, through negotiation, then accepting and achieving a new, redefined sense of normality.
A Victorian mother of a 2 year old child described how she transitioned from devastation of her
child’s diagnosis to a redefined sense of normality, ‘perhaps I was in shock. It took me a long time to
adjust to XXX's diagnosis but now it’s just kind-of normal really.’
Accordingly, the experiential basis of new normal for parents in my research - the meaning
of new normal - is that intra-psychic processes of development and negotiation have reached a point
where parents feel comfortable and confident in those redefined, newly established expectations
and identities, roles and responsibilities for treatment management; embracing a redefined reality
and adapting to their child’s diagnosis. This appears then to be a function of time and experience;
time to digest and accept the diagnosis, time to develop and establish redefined expectations, time
to reimagine, accept and embrace the parental self including role, responsibilities and identity, time
to routinise CF treatment regimen and early surveillance for CF lung disease and time to acquire
confidence and comfort in these redefined constructs through experience of them. Successful
115 Parenting Children with Cystic Fibrosis
management of these factors could represent adaptation to CF (which will be discussed in due
course).
Whilst maintaining a new sense of normality appears a dynamic process, the cross-sectional
nature of the research meant I was not able to discover whether maintaining a new normal is a
dynamic process most likely because of differing treatments, varying levels of child health, hospital
admissions and disease progression that need to be continuously merged into family, as suggested
by Hewitt-Taylor (2009) and MacDonald and Callery (2007). Stratification of child age in the my
research identified that parents of children of varying ages perceived they had achieved a redefined
sense of normality, suggesting a dynamic process of redefinition due to different events and
circumstances for these families needing to be incorporated into the family.
Bluebond-Langner (1996) attests to a dynamic process of redefinition to accommodate the
child’s condition and treatment. For example, she reported that parents were able to incorporate
their child’s first hospitalisation within the parameters of normal as they had redefined them by
renewing their efforts to establish the situation within the realm of their new normal. However, it
remains less understood whether, and how, further events such as child health deterioration and/or
increasing hospital admissions might disrupt early conceptualisations of redefined parent and family.
Parents in my research are thrust into a situation where child health deterioration and increasing
hospital admissions is a product of the early surveillance experience that may influence those early
conceptualisations of a redefined sense of reality.
In review, diagnosis of CF causes disruption in early parenthood by causing collapse in
expectations associated with parenting because parents do not know, or have little knowledge,
about CF and its management. Collapse in expectations of family life and what it means to be a
parent can result in psychological distress including frustration and guilt of having an ill child, along
with fear of the future and despair about the present. Collapsed expectations must be redefined
with CF included in a new sense of family life – new normal. Negotiating a CF diagnosis and its
treatment management into parent identity and expectations (parent and family) results in
redefinition of these components of family life. Parents of children all ages expressed their desire to
engage with other parents to gain perspective not only on clinical and genetic aspects of CF, but of
the personal experiences of CF and early surveillance. As early surveillance is generally introduced to
parents concurrently with diagnosis, they become part of the same thing. These are two sides of the
same experience. Familiarity with a redefined reality of CF and early surveillance confirms the
structure of new normal. Below is a diagrammatic representation of intra-psychic processes parents
followed to reach the outcome of a redefined sense of reality – new normal. This theme identified
both understanding and experience as mechanisms for negotiation of expectations and parent
116 Parenting Children with Cystic Fibrosis
identity. The next theme will explain how knowledge (per se and from experience) facilitates
understanding.
Figure 6. Representation of how parents psychologically reach an alternate sense of normal family
life
5.4 Understanding the unknown and understanding uncertainty
This theme describes the journey from knowing nothing or very or little about CF and early
surveillance to understanding the chronic disease and its management by AREST CF. Understanding
the unknown refers to parents’ journey from not knowing about CF and early surveillance to
understanding them. Whereas understanding uncertainty refers to parents’ understanding of the
uncertain morbidity and mortality associated with CF; because part of understanding CF is
understanding and accepting uncertainty in long-term health outcomes for their child.
Having little or no knowledge about CF is the dominant circumstance for parents in my
research and this is consistent with other studies showing that parents of newly diagnosed infants
may have heard of CF but possess no concrete understanding of this chronic condition (Massie, et al.,
2007; Tluczek, et al., 2009). Lack of knowledge about CF potentiates collapse of expectations and
previous knowledge about parenthood and family life. Therefore, understanding CF is of paramount
importance to parents because it is understanding that enables parents to start redefining self, child
and family life based on their acquired knowledge.
117 Parenting Children with Cystic Fibrosis
Essentially, it is understanding that helps parents reconstruct their collapsed life worlds. That
is, understanding is the mechanism by which parents proceed from collapse to their new normal.
Moreover, understanding CF and early surveillance facilitates belief and confidence in parents’ ability
to master their new way of life. Through the process of understanding CF and early surveillance,
parents come to understand that they can achieve their new normal. To explore the varied aspects
involved with negotiating CF and its treatment within context of early surveillance, the following sub-
themes were identified: understanding CF and understanding early surveillance of CF lung disease.
5.4.1 Understanding CF.
As parents’ expectations for their futures collapse, they are desperate to know they can
manage their situations and their children because at the point of diagnosis, they have no sense that
they are able to manage. Parents are also desperate to establish some sense of what their child’s life
may be like because collapse in those expectations and aspirations extends beyond their own and
their families’ life to the child’s life. Will they be able to play sport like other children? Will they get
married and have children? Will they be able to get a job? Projections of their children’s futures such
as these are questioned by parents during the early phase of diagnosis because of their lack of
understanding about CF. Parents seemed desperate to claw their way back to something that they
could believe and accept. This process was achieved through understanding their children’s
condition; how it could be managed, their prognosis and their quality of life. The following quote
from a Western Australian mother of a 4 year old child exemplifies how knowledge of CF and early
experiences facilitated her redefinition of expectations, ‘it has got better and that comes down to
experience and understanding. Like I said, those first few months after diagnosis, if you have no idea
that CF is even a possibility and you're child has CF, you're hit with a sledgehammer of emotion and
it's, you just, you're in a little bubble and it takes a long time for you to get out of that and I think
that's what makes the difference. You suddenly start having all of these experiences, you start
knowing about what's going on, that you start feeling a lot calmer about what's going on.’
Knowledge and experience in the early stages of receiving a child’s CF diagnosis allow
parents to begin to come to terms with their child’s diagnosis; start to make sense of it and put
boundaries around it. Knowledge and experience gives parents understanding; understanding of a
condition, a diagnosis that, because of lack of understanding, initially took everything about the
future of parenthood and family life away. Moola (2012) described how information about the
condition and experience of their child facilitated normality for the family and alleviated stress for
parents. Jessup and Parkinson (2010) reported that parents redefined their family’s expected future
through negotiating and reconstructing their current understanding of family life after it had been
threatened by a CF diagnosis by assimilating new information about CF into the expected future. That
118 Parenting Children with Cystic Fibrosis
is, knowledge about CF starts to put a limit on what a parent thinks in terms of their own, their
family’s and their child’s futures.
Accumulation of knowledge about CF frames the devastation and gives parents a foundation
to start redefining their lives and consequently giving them belief in their ability to manage their
situation. It is through understanding how CF will fit into family life, and not take over family life (as
first thought, or as actually happens as part of the diagnosis) that parents start to construct and
harbour belief in their ability to execute and achieve what is being asked of them by medical staff. A
Victorian mother of a 5 year old reflected on her transition from devastation of diagnosis to
normality when she said, ‘that's our life now, but you don’t always feel this level-headed, sometimes
you had your doubts…but that's what we've been dealt and you learn to deal with it.’ Similarly, a
Western Australian mother of a 2 year old child spoke about her transition, ‘I try not to make it a
really big deal of it anymore…we are pretty confident now that we can handle the situation but it
wasn’t always like that.’ Whether parents have belief in their ability to look after their child and to
master their new way of life from the beginning or whether it is a process over time, parents in my
research felt they had reached some level of acceptance and achievement of their new normal
through their understanding of CF and their belief to execute their new medicalised roles.
Accumulation of knowledge about CF and the experience of its management are tools for
redefining expectations about parenthood and family life. The most salient example of this process
in my research was that detailed knowledge of CF, and experience of the complex care regimen,
appeared to contribute to parents’ desires for treatment mastery and to their subsequent beliefs in
their ability to successfully conduct treatment. Therefore, whilst parents can become overwhelmed
with treatment requirements as they learn about CF care, over time they develop considerable skills
(and perceived competence) in the condition’s management (Bluebond-Langner, 1996; Grossoehme,
et al., 2014). This process and outcome have been shown in other paediatric chronic conditions
(Balling & McCubbin, 2001; Bjork, et al., 2009; Cashin, et al., 2008; Cederborg, Hultman &
Magnusson, 2011; Hewitt-Taylor, 2009; Kirk, et al, 2005; Lee & Rempel. 2011; Nuutila & Salantera,
2006; Rehm & Franck, 2000; Tong, et al., 2010). It is through gaining knowledge of the condition, and
in their ability to maintain treatment, that parents develop competence and confidence in their
abilities and judgements about chronically ill children (Balling & McCubbin, 2001; Cashin, et al.,
2008; Cedeborg, et al., 2011; Hewitt-Taylor, 2009; Kirk, et al., 2005).
Understanding and experience of a previously unknown entity are two defining factors that
provide a framework to make sense of what is unknown, so that transition to ownership and
acceptance of choices in an alternative reality can take place. A similar process was reported in
Bluebond-Langner’s (1996) ethnographic study with parents of children with CF. The study reported
that parents’ ability to manage CF-related tasks increased over the first year, which in turn gave
119 Parenting Children with Cystic Fibrosis
parents confidence in their increased mastery, creating a new routine that become normalised with
experience over time. By gaining knowledge, experience, confidence and competence in CF
treatment management, parents are likely to manage well with the daily routine associated with CF
management. From the following excerpts, it can be seen that parents progress to a point where
they have a comprehensive understanding of how the CF treatment regimen fits comfortably into
family life:
Western Australian mother, 5 year old child - Caring for of XXA has got a little bit easier.
No, I wouldn't say it's gotten easier but it's more manageable. I mean, I do manage
which provides me with the knowledge that I can manage, know what I mean.
Western Australian mother, 18 month old child - every day you’re doing 2 rounds of
physio and she’s on anti-biotics twice a day and vitamin supplements and saline
supplements but it’s just part of the routine now, so it sort-of becomes a background
thing and you just do it. When we first did it we used to get achy arms and things and
wonder how we were going to do this all the time, but now it’s just part of the routine.
Additionally, parents who expressed they regularly received assistance from outside the
home to care for their child/ren were the same parents who spoke about how they had assimilated
CF management into their daily routine, suggesting it is not necessarily only time that facilitates
adaptation to CF management. Identifying that family assistance outside of the home may help
parents adapt to their child’s CF management has been insufficiently examined (Grossoehme, et al.,
2014). However, in families where grandparents provided little or no support, an increased tendency
for parental depression has been observed (Hodgkinson & Lester, 2002).
Reflecting contemporary childcare arrangements, this line of enquiry could highlight to
parents that sourcing family assistance could help them to successfully adapt to CF management.
Due to the exploratory nature of my research, it falls short of stating causal relationships between
parental factors such as use of support and adaptation to CF management. There is, however,
evidence that social support is a modifiable risk or protective factor (McCubbin & McCubbin, 1993;
Wallander & Varni, 1998; Horton & Wallander, 2001), suggesting social support is indeed a
mechanism for adaptive coping and functioning for parents of children with CF (Szczesniak, Zou,
Wetzel, Krause & Grossoehme, 2015) and other chronic conditions (e.g., Hoekstra-Weebers, et al.,
2001; Kars, et al., 2008; Khurana, Katyal & Marwaha, 2006; Spiers, et al., 2011).
Determining whether family assistance and other similar support structures contribute to
mal/adaptation to a CF diagnosis and to the unique CF management of AREST CF are important steps
in identification of targeted approaches that can assist parents in adapting to CF management in the
context of early surveillance. Moreover, implementing targeted approaches that assist adapting to CF
120 Parenting Children with Cystic Fibrosis
management may also be conducive to parents’ negotiation of their new normal within context of
early surveillance.
If we now turn to the latter part of the theme title understanding uncertainty, one of the
fundamental aspects of understanding CF is acknowledging the characteristic variability in morbidity
and mortality (i.e., life expectancy). Whilst there is an average life expectancy of 37 years for females
and 40 years for males (MacKenzie, et al., 2014) , long-term health outcomes for individuals with CF
are variable (Rowntree & Harris, 2003; Zielenski 2000). Therefore, understanding uncertainty refers
to the matter of understanding uncertainty in terms of morbidity and mortality as a characteristic of
CF: it refers to how uncertainty in their child’s life, and its associated fear, becomes a lived
experience for parents of children with CF.
As part of understanding CF, parents must acknowledge and eventually accept that life
expectancy and morbidity are variable; understanding the uncertainty, understanding there will be
an element of unknown. Understanding uncertainty in prognosis causes fear about the present and
the future (Afonso, Gomes & Mitre, 2015; Havermans, et al., 2015; Moola, 2012). Once a child is
diagnosed with a chronic condition, parent’s fear about their future is a salient finding in the
literature (Arestedt, Persson & Benzein, 2014; Gjengedal, et al., 2003; Moola, 2012; Patrick-Ott,
2011). Fear about a child’s future is supported by Jessup and Parkinson (2010) who reported parental
fear at diagnosis and then ongoing fear as part of parenting a child with CF. A lived experience for
parents of children with CF is living with uncertainty and fear.
Living with uncertainty is demonstrated by a Western Australian mother of a 2 year old child
who said ‘sometimes I feel like I’m waiting for the shit to fall because to look at her, you wouldn’t
know that she is a sick kid…it’s like we know, I know, that it’s going to happen at some point…I’m still
waiting for that. It can be a really scary prospect, you know.’ The sentiment was echoed by a
Victorian mother whose child was 4 years old when she stated ‘she knows what CF is, she doesn't
know she's going to get sicker, she doesn't know she's going to die at a young age. I’m still dealing
with the fear of that happening and so I don’t want that on my daughter just yet.’
For parents in my research, the uncertainty surrounding morbidity and mortality are a source
of fear that likely goes beyond general parenting levels. Descriptions are similar to those reported by
parents of other chronic (Cabizuca, Marques-Portella, Mendlowicz, Coutinho & Figueira, 2009;
Patrick-Ott, 2011; Zhou, et al., 2014) and life-limiting conditions (Greening & Stoppelbein, 2007;
Hoekstra-Weebers, et al., 2001; Kazak, et al., 2005; Von Essen, et al., 2004). Following diagnosis,
redefined expectations and indeed, new normal always contain a level of uncertainty around a child’s
morbidity and mortality, thereby inherently eliciting some level of fear.
Uncertainty in Illness theory (Mishel, 1990) presents a basis for understanding how
uncertainty associated with chronic illness is experienced. There are multiple aspects of uncertainty
121 Parenting Children with Cystic Fibrosis
in illness covered by Mishel’s theory, but the inability to accurately predict outcomes of the
condition for the inflicted person is particualry relevant to my research. Uncertainty reflects a
neutral cognitive state and is neither desired nor dreaded until the implications of uncertainty are
determined. That is, individuals appraise the situation and determine the value placed on
uncertainty. Parents in my research decided whether uncertainty surrounding morbidity and
mortality associated with their child’s CF was a positive or negative event within their redefined
expectations. Uncertainty in the child’s condition was primarily viewed as a negative event that
induced fear.
Fear was for the unfamiliar present and uncertain future. Fear of the present was about
current logistics of disease progression, including potential hospital admissions, extra treatment,
care for siblings, and time off work. Fear of the future concerned fear of losing the child prematurely
and fear of the child’s health deterioration. Moola (2012) and Jessup and Parkinson (2010) described
how parents of children with CF constantly worried about child health deterioration, conceptualised
as fear by the authors of both studies. Parents of children with CF can carry a level of fear that can
overcast, but may not significantly affect, parenthood and family life.
Whilst uncertainty in morbidity and mortality in most cases is initially perceived negatively by
parents, and certainly influences parental fear, the potential variability in long-term health outcomes
for people with CF actually provides means for hope for a child’s future. To explain further, parents
of a child diagnosed with a disease or disability without potential for variability in outcomes (for
example, parents of a paraplegic child) could be less likely to embrace hope for positive long-term
health outcomes. Whereas for parents of a child diagnosed with a chronic disease such as CF that has
the potential for variable long-term health outcomes, the negativity that surrounds uncertainty in
morbidity and mortality can be surpassed by hope for their child’s future. Hope for a child’s future is
elaborated in the next chapter as an over-arching lived experience for parents in my research.
Mishel’s (1990) theory explains that uncertainty in illness can be perceived as a positive
event when individuals assign beneficial value to the uncertainty. That is, whilst diagnosis of CF
cannot and should not be patronised into a beneficial event, when parents in my research assigned
positive value to uncertainty, they were able to construct psychologically beneficial perceptions of
their child’s condition. Favourable perceptions from assigning positive value to uncertainty resulted
in constructions of hope for the child’s future in terms of quality of life (i.e., morbidity) and life
expectancy (i.e., mortality). Therefore, remarkably, there appears to be an evolution, a
reinterpretation (though there can be some regression), in the way uncertainty of morbidity and
mortality is constructed.
This finding supports Mishel’s (1990) conceptualisation of how cognitive appraisal and
assignment of value influences constructions of uncertainty in illness. The finding also supports
122 Parenting Children with Cystic Fibrosis
Lazarus’s and Folkman’s (1984) early theory about the role of appraisal in coping with stressful
events, in that appraisal of an event, and assignment of value to that event as a result of the
appraisal impacts upon cognitive and emotional constructions of that event, thereby contributing to
coping resources drawn upon. How cognitive appraisal of an event influences choice of coping
resources and strategies will be the topic of discussion in the next chapter. The following quotes
exemplify how some parents constructed perceptions of uncertainty in their child’s condition in a
positive manner:
Victorian father, 12 month old baby - You know, I can't control it so at the end of the day
I'm not going to stress over something that I can't control…There's the other side too,
some people have a fear of it getting worse. But it's like 'ok, nothing may happen.' So
we’ll just keep doing what we’re doing and that, and away you go.
Western Australian mother, 4 year old child - in my head I always think that if you’re
going to think the worst then contemplate the best part as well…If you’re going to sit
there and say that this and this could happen, then you’ve also got to think that this and
this could happen.
Therefore, parents’ dynamic journey of learning about CF transforms them from devastation
of diagnosis to understanding of CF – understanding the unknown and understanding there is
uncertainty. Moreover, the uncertainty of a child’s morbidity and mortality can be over-arching and
all encompassing, with either positive or negative effects.
5.4.2 Understanding early surveillance for CF lung disease.
Similar to the lack of knowledge about CF at time of diagnosis, parents also lack knowledge
of early surveillance. They lack understanding of the processes and procedures, and of their own
experience. The procedures and their purposes are comprehensively explained to parents, but there
are elements of early surveillance that are not, and cannot be, fully explained by researchers or
clinicians. These elements are especially prominent for the first annual early surveillance review, but
there remain elements of uncertainty in all annual reviews. Elements of early surveillance that
remain uncertain during the first annual review are the substance of this sub-theme, and how they
evolve to being known. Whilst explanation of early surveillance processes and procedures are greatly
appreciated, parents are uncertain about their own personal experience (i.e., how their child will
respond and their own psychological experience) until such time when they experience the
program’s components and subsequently gain further knowledge.
Around the time of diagnosis, unknown parental experiences and child responses to early
surveillance procedures add to parents’ feelings of uncertainty about the present and future; parents
are not only negotiating redefinition of self, family and future expectations. Parents’ first experience
of early surveillance is generally closely paired with CF diagnosis. Consequently, the first early
123 Parenting Children with Cystic Fibrosis
surveillance review appears to continue or exacerbate similar thoughts and feelings to that of
diagnosis. This is not to diminish the heart-wrenching experience of diagnosis but to demonstrate the
potential follow-on effects, or likely exacerbation, of emotions at the first annual review. Qualitative
studies exploring the lived experience of parenting a child with CF have reported how events such as
required surgical procedures for a child can trigger thoughts about CF (Bluebond-Langner, 1996;
Glassoce & Smith, 2011). Likely running concurrently with diagnosis, parents conceivably need to
negotiate through their first early surveillance experience, which will eventually become part of their
new normal. The following quote demonstrated how a Western Australian mother of a 4 year old
child felt about her infant’s first annual review in relation to diagnosis:
You’re still getting to grips at that point with the diagnosis, because we went through
the denial thing, you know “are you sure you haven’t mixed up the tests?” that sort-of
thing…so you still haven’t really accepted it at 12 weeks, you still haven’t accepted that
your child has CF and that your life is changed…as I said, that first time, you are still...you
know, you're still coming to grips with the diagnosis.
In terms of parents’ perceptions of the received level of information about early surveillance,
some parents found the level of information overwhelming. A Victorian mother of 4 year old child
reflected on her experience of receiving information about early surveillance, ‘it goes straight over
your head because they barrel you with all this information, new procedures, new everything, and it
doesn't quite sink’. Other parents felt information they received was adequate. A Western Australian
mother of a 5 year old child recalled ‘I think that the level of information that we're given is
good…you know, lots of information prior, so I've been really happy with all of that.’ Whereas other
parents felt the information they received about early surveillance was insufficient for an informed
decision, reflected by a Western Australian mother’s account whose child was 4 years of age, ‘I don't
think we really understood what the tests were for and what the outcomes were going to mean so we
felt that we could’ve made an uninformed decision.’
Jessup and colleagues (2015) found that most parents’ were overwhelmed with the amount
and diversity of information at the time of their formal CF education, whereas Sawyer and Glazner
(2004) described parents’ satisfaction with the amount and content of information given during CF
management training. Whilst these limited results reflect diverse parental perceptions of Australian
standards of care, they do not represent information given beyond formal CF education and are
therefore not generalisable to parents in receipt of information about early surveillance for CF lung
disease. It is conceivable that any confusion about early surveillance may lead to perceptions of
insufficient information, miscommunication or indeed, the therapeutic misconception (National
Bioethics Advisory Commission, 2001), explained in detail in a later theme. Therefore, findings from
124 Parenting Children with Cystic Fibrosis
my research suggest that protocol development around information content and delivery is
warranted, and could inform development of such a protocol. At the very least, medical staff can be
cognisant of parent information needs and priorities, both around the time of CF diagnosis and more
specifically, about early surveillance.
There is one form of communication about early surveillance that parents felt was lacking.
This information is about their potential personal experiences of the days’ proceedings; how they
might manage with the day, how they might feel, their responses and reactions. There is no advice or
illumination of the potential psychological experience that parents may encounter throughout their
day. Consequently, for parents with no annual surveillance experience, there is no foundation, no
boundaries to confine and control their anticipatory ideas and notions about the day’s events.
No foundation or boundaries for what parents’ may experience speaks to the unknown
conceptualisation of early surveillance in multiple ways; unknown about their child’s reaction,
unknown of their own reaction, unknown about the staff who will disappear with their child,
unknown about whether their child will be relinquished back to them, unknown about their child’s
prognosis and unknown about their child’s longevity and quality of life. This creates a lack of
expectations that could be mitigated, or at least reduced, if there was some kind of notification of
what parents’ personal experience may be like; a kind-of foundation or framework upon which to
base and confine their anticipations.
Applicable to parents of younger children (especially first-time parents) are the psychological
consequences of not possessing a frame of reference because of lack of expectations for their
ensuing personal experience of annual review. No foundation or framework for expectations is the
consequence of the unknown; similar to lack of expectations at the time of diagnosis. This
phenomenon is demonstrated through a Victorian mother’s (child aged 2 years) experience who had
no assumptions of, and therefore lack of expectations about, early surveillance and how this affected
her first experience, keeping in mind how closely paired to diagnosis the initial visit generally is:
When he was really small, I found it harder because you didn’t really know what to
expect. I didn't know how he was going to deal with it, I wondered if I was doing the
right thing because he had to have things done to him that he otherwise wouldn’t have
to…I cried when he was a baby because it was all new and very hormonal and still
unsure. You're trusting these people that you've never trusted before with your baby
but now, having done it a few times you know that they do a good job.
It is worthy to note here that parents with substantial early surveillance exposure (that is,
parents of children aged 3 years and older) expressed their willingness to consult with parents new
to early surveillance. They also asserted their belief that such communication could promote more
rapid adaptation to both diagnosis and early surveillance than their experience. A Victorian father of
125 Parenting Children with Cystic Fibrosis
a 5 year old child asserted, ‘I think it would be good for people who are coming into that position to
talk to other families, or if there was a network of families that you could talk to and they could tell
you what’s going on and what’s to be expected. It wasn’t until afterwards that someone spoke to me
about that stuff, about what’s happening, what you’re feeling is normal while going through that so I
think it would’ve really helped us at that time.
Parents with children under 3 years of age spoke about their desire to communicate with
parents about their experiences of early surveillance - of their thoughts and emotions - with the hope
of being more prepared for their first early surveillance exposure. A Western Australian mother of 2
year old child stated, ‘it would also be good to get information from other parents, you know like how
they get through the bronch (bronchoscopy). It would be good to get just little snap-shots about what
will happen and how they dealt with it. Even just things like, from a parent’s perspective watching
your child go under general anaesthetic, this is what happened and this is how I dealt with it...not to
dramatise it but just to bring it to people’s attention. Although everyone's experience is different, at
least to have some idea of what it might have been like would’ve been good.’
To summarise, after collapse of parenting expectations and dismantling of parent identity,
parents come to understand how to manage their child’s CF through establishing expectations about
CF management (including family routine) and prognosis. Knowledge of these entities gives parents
a foundation to start reconstruction. An aspect of CF that becomes understood by parents is the
variability in morbidity and mortality, which contributes to the lived experience of uncertainty and
fear about child, family and their own future. However, the uncertainty in mortality can also result in
hope for their child’s future because although a clock has been set on their child’s life, the variability
is constructed as an avenue for hope.
As early surveillance is inextricably linked to CF, it becomes part of the lived experience of CF.
The link between CF and early surveillance may continue or exacerbate psychological distress
experienced at diagnosis, and could indicate psychological adversities and processes experienced by
parents whose children undergo early surveillance for CF lung disease. An implication for parental
outcomes as a result of their child’s first early surveillance is that parents are willing to talk with
other parents about their experiences so that they can have an idea of what will happen and how
they might respond on a personal level. It is through experience and subsequent knowledge of their
ability to manage the day’s event and their child and their own responses that parents become
comfortable and feel they can cope. It is through understanding, that parents come to accept their
child’s diagnosis and early surveillance of CF lung disease as part of the redefined expectations and
reimagined parent identities. Discussion will now shift to the everyday regular routine of parenting
children with CF in the context of early surveillance.
126 Parenting Children with Cystic Fibrosis
5.5 Good days and bad days; Fluctuation between positive and negative outlook on life
Most of the literature to date examining experiences of parenting children with CF has
evaluated the diagnosis phase (Armoni, et al., 2008; Priddis, et al., 2010; Carpenter & Narsavage,
2004; Sawyer & Glazner, 2004; Tluczek, Koscik, Farrell & Rock, 2005) and its resulting
psychopathologies (Glasscoe, et al., 2007; Goldbeck, 2006; Merelle, et al., 2003). Fewer studies have
assessed parental outcomes beyond the diagnosis period (Delelis, Christophe, Leroy, Vanneste &
Wallaert, 2008; Gjengedal, et al., 2003; Jessup, et al., 2015; Tluczek, Clark, McKechnie & Brown,
2014), including examination of positive parental outcomes such as psychological growth (referred
to in the literature as resilience and post-traumatic growth) (Cadell, et al., 2014, Nabors, et al., 2013;
Phipps, et al., 2015).
This theme is an all-encompassing description of parental experience beyond diagnosis, that
is, the day-to-day experience of parenting a child with CF within the context of early surveillance.
Therefore, this theme adds to the limited knowledge about parents’ experiences of CF and its
management beyond diagnosis, and demonstrates how early surveillance contributes to these
regular, day-to-day experiences. Taken together, this theme describes the lived experience of
parenting a child with CF in the context of early surveillance.
The current theme draws upon limited literature as a contribution to how parents construct
meaning around their day-to-day experience of their child’s CF beyond diagnosis. The first two sub-
themes detail parents’ lived experience by presenting salient psychological constructs that exist on a
daily basis. The final sub-theme elucidates parents’ fluctuation between these two perspectives as
their lived experience of CF. Overall, this theme asserts that for parents in my research, some days
are good and some days are bad in terms of their perspectives on CF, and that parents fluctuate
between their positive views and emotions and their negative views and emotions. This theme
relays these accounts through identification of three sub-themes: bad days, good days; and
fluctuation between positive and negative outlook on life.
5.5.1 Bad days.
Adverse outcomes for parents of children with CF, and for parents of children with other life-
limiting and chronic conditions, have been generally expressed in the literature as caregiver burden,
treatment burden and parenting stress (Aydinok, Erermis, Bukusoglu, Yilmaz & Solak, 2005; Brod,
Hammer, Christensen, Lessard & Bushnell, 2009; Couzino & Hazen, 2013; Hall & Graff, 2011; Hassall,
et al., 2005; Henry, et al., 2008; Hutchinson, et al., 2009; Klassen, et al., 2010; Litzelman, et al., 2011;
Norberg & Steneby, 2009; Quittner, et al., 1992; Sav, et al., 2013). Similar to other studies, parents in
my research described ensuing adverse psychological outcomes beyond diagnosis (Goldbeck, Besier,
Hinz, Singer & Quittner, 2010; Quittner, et al., 2008; Quittner, et al., 2014). Stratification of my
sample by child age identified that parents in all age groups expressed such effects, suggesting that
127 Parenting Children with Cystic Fibrosis
adverse psychological outcomes begin with the destruction of CF diagnosis (Glasscoe, et al., 2007;
Goldbeck, 2006; Priddis, et al., 2010) and ensue thereafter. Adverse parental psychological outcomes
comprise the first part of this theme; fear and chronic sorrow.
5.5.1.1 Fear as an outcome of uncertainty in morbidity and mortality.
Parental psychological adversity experienced on bad days primarily consisted of fear, which is a
natural response to general life events (Olsson & Phelps, 2007). Whilst fear can be positive in limited
levels because it can protect and motivate (Ulrich, 1983), it appeared to go beyond these limited
levels. Therefore, general parental fear is exacerbated for parents of chronically ill children (Crnic &
Low, 2002; Krockek & Mowder, 2012). This is not a new finding in parents of children with CF (Fidika,
et al., 2015; Hodgkinson & Lester, 2008; Jedlicka-Köhler, et al., 1996; Jessup, Douglas, Priddis,
Branch-Smith and Shields, 2015; Wong & Heriot, 2008). However, due to the unique nature of early
surveillance, how it contributes to the presence of parents’ fear has not been previously explored.
Fear is generally caused by a perception of risk; it is an affective response of unease or
apprehensiveness to a situation that has been cognitively appraised as dangerous (Lerner & Keltner,
2001). Though parents do not perceive themselves in an immediately threatening, or risky, situation,
it is the unknown consequences of CF that are the drivers of fear, that is, the uncertainty in morbidity
and mortality identified in the previous theme. Parents experience fear for this reason intermittently,
on what they expressed as their bad days. There are a number of dimensions to the unknown
consequences of CF that generate fear. Firstly, fear of losing a child to CF was expressed by a
Victorian mother of a 6 month old baby who said ‘sometimes I have a constant worry in the back of
my head that something might be wrong, that fear of losing my child. I think ‘why us, why her.'
Secondly, fear of a child getting sick due to disease severity was described by a Western Australian
mother of a 2 year old child who stated ‘so far, so good sort-of thing, but there’s still that element of
stress. That’s the other thing, because XXA has been so well, sometimes I feel like I’m waiting for the
shit to fall because to look at her, you wouldn’t know that she is a sick kid…It’s like we know, I know,
that it’s going to happen at some point but we’re still waiting for that, I’m still waiting for that.’
Lastly, fear of knowing a child is getting sick through knowledge of early surveillance results was
expressed by a Western Australian father whose child was 18 months old when he said, ‘you don’t
know how the results really fare compared to just being nice and clean. That’s what I sort-of really
worry about.’
Self-management of psychological adversities is important for parents’ well-being and that of
their family members (Ackard, Neumark-Sztainer, Story & Perry, 2006; Whittemore, et al., 2013).
Several strategies were identified that enabled parents to manage (or cope with) their fear that was
associated with uncertainty in morbidity and mortality. Firstly, some parents spoke about their
psychological efforts to eliminate or minimise any adverse thoughts or feelings (Fidika, et al., 2015;
128 Parenting Children with Cystic Fibrosis
Sheehan, et al., 2014). For example, parents tried not to think about their child’s condition in such a
way that it affected them psychologically. This is represented by a Western Australian mother of a 6
year old child who tries to ‘push it all the way down, right down to the bottom of your feet and you
don’t think about it. Just, you’ve got to block it out, because if you do think about it, and we have at
times, you get consumed by it, it just eats you up. So you’ve just got to, I think, block it out.’
Secondly, parents expressed their desire and ability to release their emotions periodically
during such times that warranted it (Coyne, 1997; Tong, et al., 2010; Venters, 1981; Zubrzycka, 2015).
In this instance, parents would get angry or cry, which diffused their emotional burden. For example,
a Victorian mother of a 5 year old child spoke about this process when she said, ‘that's our life now
but you don’t always feel this level-headed, sometimes you have your down times…I just let myself
have a good cry. I just allow myself to have those moments.’
Thirdly, parents restructured, or reconstructed, their thoughts to reframe their situation
within context of the wider society (Havermans, et al., 2015; McCubbin, et al., 1983; Venters, 1981;
Zubrzycka, 2015). This refers to parents’ strategy of comparing their life situation to those who have
to manage worse situations, described aptly by a Western Australian father of a 2 year old child who
said, ‘the only thing that helps me stay sane is that you see things on TV about kids every night who
don’t have access to medical care and doctors and things like that. The way I stay sane is I think - how
lucky are we to have access to all this.’ Whilst this does not change their situation, it changes parents’
cognitive constructions of it. This has been referred to in psychological literature as social
comparison (Festinger, 1964; Wills, 1981), and this concept will be elaborated upon later.
Fourthly, support was a strong resource for coping with fear (Coffee, 2006; Green &
Stoppelbein, 2007; Havermans, et al., 2015; McCubbin, et al., 1983; Priddis, et al., 2010; Zubrzycka,
2015). Support was mainly used as an avenue to reduce psychological and/or emotional burden
either with a partner, family or friends (Wong & Heriot, 2008); and was reported more often by
mothers compared with fathers. A mother from Victoria whose child was 5 stated, ‘just dealing with
it together, having a support person is wonderful, it really helps me,’ and the sentiment was echoed
by this Western Australian mother whose child was 6 years old when she described, ‘ just to kind-of
talk about it with friends and family makes it easier.’ This finding is in agreement with Havermans, et
al., (2015) who illustrated that mothers used social support as a coping resource more often than
fathers.
Finally, staying in the present moment and not letting thoughts go too far into the future
helped parents to manage their fear about uncertainty that has been built into their futures as part
of that redefined new normal (Gjengedal, et al., 2003). To illustrate the point, a Victorian mother of a
12 month old baby asserted, ‘there's no sense in lulling over what could possibly go wrong in the
future, it may or may not even happen, you just deal with what is in front of you now.’ Discussion of
129 Parenting Children with Cystic Fibrosis
theoretical application and wider implications of some of these coping strategies, along with how
they might apply to context of family life, occurs in the next chapter.
From the above quotes, coping with fear associated with CF is primarily represented by an
emotion-focussed coping approach (Lazarus & Folkman, 1984). Recall from Chapter 2 the
Transactional Model of Stress and Coping (Lazarus & Folkman) where emotion-focussed coping
comprised a major component of how people choose to cope with life stressors. Emotion-focussed
coping strategies are primarily utilised by parents in my research because, through their appraisal,
they understand they cannot remove the stressor of CF, and so come to use strategies that enable
regulation of emotions associated with it. For example, Lazarus and Folkman’s psychological
conceptualisation of coping with fear applies to the processes followed by parents in my research,
that is, appraisal of an event as threatening induces fear, and fear induces utilisation of available
coping resources dependent on the appraisal and on the availability of coping resources for the
individual.
A number of parental characteristics for coping with fear were discovered. Firstly, parents
whose children were 3 years of age or older often spoke about how they managed their fear,
suggesting that developing skills to manage fear associated with CF and/or early surveillance may
take time and experience. Secondly, parents who had access to external assistance for their children
spoke about their ability to manage their fear, suggesting that resources outside of the nuclear family
help parents to cope. Recall that family assistance facilitated the normalisation process as part of
adjustment to diagnosis. These findings highlight importance of extended family and external
networks as a continual source of support that enables parents to better function in their roles as
carer, parent, and likely within themselves. Thirdly, full and part-time working mothers more often
spoke about effectively coping with fear than mothers who classed themselves on full-time home
duties. This particular finding is in agreement with Carver (1997), who found that working mothers
coped more effectively with life stressors than mothers who did not work.
Collectively, these findings suggest there is potential for outside influences to contribute to
parents’ coping resources. For example, more effective coping by working mothers versus stay-at-
home mothers may occur because time outside of the immediate family environment could function
as a reprieve for mothers, resulting in giving them more scope to deal with personal adversity
(LeMaster, Marcus-Newhall, Casad & Silverman, 2004). Parents may learn coping skills outside of the
family setting either from managing work-related issues or through talking with people outside the
family home (Gatrell, 2013). Finally, parents who spoke about coping with fear had older children,
suggesting the possibility that generic parenting skills developed before birth of the child who has CF
may buffer against fear.
130 Parenting Children with Cystic Fibrosis
Parental fear of disease progression is a recent line of inquiry examining existential outcomes
of parenting a child with CF. Fidika and colleagues (2015), Jessup and Parkinson (2010) and Wong and
Heriot (2008) examined fear of disease progression and/or morbidity and mortality. Together, these
studies reported an ongoing process of fluctuating fear that affects parents’ adjustment to their
child’s CF. Given that parental fear of disease progression in CF is a fairly new concept in the
paediatric chronic illness literature (Fidika, et al., 2015) and that early surveillance of covert lung
disease is unique to AREST CF, what is needed is investigation of how parents cope with knowledge
of a child’s disease progression. My research did not uncover if and how parents cope with such
knowledge. Given that early diagnosis and early surveillance of paediatric disease are modern
technologies, and my research uncovered the multi-dimensional nature of fear about early disease
progression, a further study with focus on how parents cope with such information and their
associated emotions and cognitions is warranted.
5.5.1.2 Chronic sorrow.
With parents’ knowledge of paediatric chronic illness comes profound and extensive sadness.
Profound and extended sadness, conceptualised in the literature as chronic sorrow (Gravelle, 1997;
Olshansky, 1962), is a prevalent experience for parents during the diagnosis phase of many chronic
conditions, including cerebral palsy, type 1 diabetes, Down’s Syndrome, physical disability and sickle
cell disease (e.g., Bowes, et al., 2009; Damrosch & Perry, 1989; Masterton, 2010, Neilsen, 2013;
Nikfarid, et al., 2015; Paul, et al., 2014; Patrick, 2014; Whittingham, et al., 2013). Beyond diagnosis, a
portion of parents are reported to experience chronic sorrow on a more permanent basis (Kearney &
Griffin, 2001; Nikfarid, et al., 2015; Patrick-Ott, 2011; Whittingham, Wee, Sanders & Boyd, 2013).
Neither grief nor loss were explicitly discussed in relation to their child’s diagnosis (as this was
not the focus of interviews), though it is conceivable that parents in my research may have
experienced this kind of profound sadness following their child’s CF diagnosis (Moola, 2012; Priddis,
et al., 2010; Shumaker, 1994). That chronic sorrow has been interpreted as an essential dimension of
the lived experience for parents in my research, it is conceivable that chronic sorrow may be a
continuation of grief or loss experienced at diagnosis. Though continuation of chronic sorrow from
diagnosis is speculative, parents in my research expressed profound and intermittent sadness
throughout their lives beyond their baby’s diagnosis, which they experienced at different times and
for different reasons. Therefore, it has been interpreted as chronic sorrow because the
conceptualisation of profound sadness in my research is comparable to the definition of chronic
sorrow described in the literature as outlined in chapter 2.
Parents’ profound sadness was about their child’s disease and their family situation. The
recurring nature of chronic sorrow is reflected within this theme because, as will be described later,
parents have fluctuating psychological states. Chronic sorrow is experienced intermittently
131 Parenting Children with Cystic Fibrosis
throughout parents’ lives. Though understood as common amongst parents of children with CF
(Shumaker, 1994), possible effects of chronic sorrow on parents and their families have not been
sufficiently studied, nor have reasons why chronic sorrow may persist beyond fear of disease
progression and loss of the child. My research adds to the scant body of literature by identifying
reasons beyond loss and grief as objects of chronic sorrow.
Firstly, parents’ expectations of a typical parental role, as well as expectations for their child
and their family, could no longer be met. In the theme redefined expectations and reimagined
identity; redefined reality, this process occurred as part of diagnosis when expectations for a typical
child and family life were destroyed. These reasons for grief and loss at time of diagnosis have been
reported in the CF literature (Hodgkinson & Lester, 2002; Hymovich & Baker, 1985; Jessup &
Parkinson, 2010). That loss of expectations was identified at diagnosis in my research, and the
literature shows both grief and loss of expectations at diagnosis can result in chronic sorrow, adds
credibility to the speculation that parents in my research experienced chronic sorrow.
If the onset of chronic sorrow did indeed arise at diagnosis, then it appears the response to
loss of parenting expectations continues intermittently. That is, on some days parents cognitively
revisit loss of expectations and at the same time, grieve for the loss of their ideologies. It is the loss
of, and grief in, losing ideologies that results in feelings of profound sadness. A Victorian mother of
12 month old baby expressed sadness about her loss of parent and family expectations when she
stated, ‘there’s time I get upset, it's hard only because I'm used to working, socialising and being out
there so it's difficult. I mean, it's not the worst thing in the world but it's just a real life-change really.
A real difference in what you expected of parenting and family life, you know.’
Secondly, parents could experience chronic sorrow when they thought about their child’s loss,
that is, the loss of quality of life and the potential loss of life expectancy. Parents felt sadness for
their child’s endurance of daily treatment, regular hospital visits, current sickness as well as their
future losses. The following excerpt from a Western Australian mother of a 5 year old child aptly
demonstrates parents’ expressions of sadness for their child’s loss, and endurance, of CF:
it’s sad because it’s not how you envisaged your child’s life to be. You always want a
healthy child and unless they had a broken arm or something, then you would never have
to go to hospital. I get upset because she doesn’t like it and I make her do things that she
doesn't like…and the pain and stress associated with it.
Collectively, fear and chronic sorrow are adverse psychological outcomes of parents’
meanings attributed to their child’s CF. That is, parents learn about, and come to accept,
characteristics of CF as they construct meaning around it for themselves, their child and their
individual and collective futures. The result of this is fear and chronic sorrow. Parents coped with
these cognitions and emotions in various ways, likely with varying degrees of effectiveness. Parents
132 Parenting Children with Cystic Fibrosis
who have; older children in early surveillance, other children, access to childcare assistance outside
of the home, supportive networks and mothers who worked at least part-time expressed coping
strategies they used to deal with their personal, intrinsic adversities, or that appeared as implicit
coping resources. Implications of coping, along with theoretical application of coping strategies and
styles will be outlined in the next chapter.
5.5.2 Good days.
Whilst parents may always carry a level of sorrow and fear about their child’s life trajectory,
there are good days amongst the bad days; but what does it mean to have a good day and what does
it feel like for parents in relation to their child’s CF in context of early surveillance? This sub-theme
demonstrates what parents experience on what they expressed as good days, comprising parental
satisfaction from perceived parenting competence, gratitude, and perceived control and power.
These ensuing beneficial outcomes of managing and living with their child’s CF, within context of
early surveillance, are reported in the literature for parents of children with CF, and for parents of
children with other life-limiting and chronic conditions, collectively expressed as resilience and
adaptation (Goldbeck, et al., 2014; Van Riper, 2007; McCubbin, et al., 1996). Research has trended
towards investigating such beneficial consequences of paediatric chronic illness for parents,
portraying resilient parents, and whether and which parent and family characteristics may contribute
or impede (e.g., Barakat, Alderfer & Kazak, 2006; Goldbeck, etal., 2001; Hullman, Fedele, Molzon,
Mayes & Mullins, 2014; Peer & Hillman, 2014; Phipps, et al., 2015). It is relevant to note that parents
in my research also experienced ensuing hope for their child’s future, but because of its centrality to
the lived experience, it is discussed as a construct that transcends both CF and early surveillance later
in the chapter.
5.5.2.1 Parenting competence supports the parental role.
The parental role refers to beliefs and values about what it means to be a parent, and
subsequent behaviours resulting from the belief and value system (Simon, 1992). Those parenting
responsibilities and activities performed to keep a child healthy (e.g., feeding, clothing and
protecting children from harm) begin to define the parental role and identity. To apply this
conceptualisation of parental role and identity to the complexity of parenting a child with CF, a
fundamental requirement for parents is to keep their child healthy by performing the rigorous and
demanding home treatment regimen outlined in the first chapter. In this sense, treatment
management becomes part of the parental role: parents’ role is to care for their child beyond typical
parenting responsibilities (as described in sub-theme Collapsed and Redefined Expectations).
The caring role often dominates the parenting role because of the need to provide ongoing
intensive care to the child (Kirk, et al., 2005; Sullivan-Bolyai, Rosenberg & Bayard, 2006). This then
133 Parenting Children with Cystic Fibrosis
contributes to perceptions of the parent as medical carer for the child, which then becomes part of
parental identity: parents become medicalised (Glasscoe & Smith, 2011, Tong, et al., 2010), and
pseudo-paediatricians (Gjengedal, et al., 2003). On days when parents feel positive about their
child’s CF, their parental role in relation to their child’s treatment management was viewed in a
positive manner. A Victorian mother of a 12 month old baby expressed how she viewed herself
when she said ‘on the good days I feel like I’m doing the right thing with her treatment so that's the
positive I get out of it as a parent, that I’m doing the best I can to protect my child’ Therefore, home
treatment management becomes inherently linked to parental identity because of intensity and
consistency of treatment, along with perceived ability, that is, self-efficacy, to keep the child healthy
- their innate parental responsibility.
In context of my research, high parenting self-efficacy includes parents’ beliefs about their
ability to positively influence the health and development of their child especially in relation to their
condition (Coleman & Karraker, 2000). The most plausible reason for high self-efficacy for treatment
is that most CF treatment is conducted within the home, and is therefore primarily the responsibility
of parents. The intensity and consistency of treatment, along with the innate responsibility to keep a
child healthy meant that parents believed they could keep their child healthy through maintaining
treatment. The central role of home treatment not only medicalises the parent role, but it places an
additional burden on the parents to achieve a degree of efficacy in delivering the treatment. If they
achieve it, the child will do better in their minds. Therefore, the child’s health outcome is intrinsically
linked to the parents’ ability to provide home treatment. Due to the centrality of the treatment
regimen to child and parent, parents in my research attributed parenting competency to achieving
the required standard and level of home treatment.
Therefore, home CF management appears a primary mechanism for achieving, and
assessing, parenting competence. Perceived parenting competence results in satisfaction in the
parenting role (Angley, Divney, Magriples & Kershaw, 2014; Bornstein, 2003; Johnston & Mash,
1989). Satisfaction in the parenting role from competence in parenting responsibilities supports
parental identity, and support for parental identity can increase parental self-esteem (Mouton &
Tuma, 1988; Ohan, Leung & Johnston, 2000). That is, playing a major role in their child’s daily
treatment management (responsibility) helped to support parents’ belief that they were doing
everything they could for their child (self-efficacy), which then gave them the satisfaction of doing so
(perceived competence). This contributes to parental beliefs (parental role) and values (self-esteem),
and what it means to be a parent of a child with CF (parental identity). The diagram on the following
page outlines how these parental psychological constructs function together and affect each other in
relation to home CF treatment and parental outcomes.
134 Parenting Children with Cystic Fibrosis
Figure 7. How parental psychological constructs are associated with home treatment and parenting
outcomes
The diagram shows that it is support for, and promotion of, self-efficacy through perceived
parenting competence that has an effect on parental identity and self-esteem. Promotion of self-
efficacy interacts positively with self-concept, that is, support of high self-efficacy promotes positive
beliefs about oneself (Fenning & May, 2013; Martin, Goldwasser & Harris, 2015). A plethora of
135 Parenting Children with Cystic Fibrosis
psychological literature supports the interaction between perceived parenting competence and self-
esteem (Gilmore & Cuskelly, 2009; Ohan, Leung & Johnston, 2000; Ponomartchouk & Bouchard,
2014; Ramassini, 2000; Rogers & Matthews, 2004). The following excerpt from a Western Australian
mother of a 12 month old baby highlights how her self-esteem was enhanced through beliefs in
parenting competence because she was able to achieve her child’s home treatment regimen, ‘if
we're not doing it, because her treatment is at home, it's in our hands. We're the ones giving her the
antibiotics and doing her physio so we are the day-to-day treatment for her, and because it’s such a
large responsibility, we feel good that we can maintain it for her.’
In conclusion, parents can experience parental satisfaction, parenting competence and
increased self-esteem on days when they have a positive outlook on their child’s CF, both in relation
to conducting and achieving their child’s home treatment as well as managing and achieving their
new normal. Therefore, managing CF and its treatment is both difficult and rewarding for parents in
their redefined roles as parents of a child requiring extensive home treatment. Consideration of
parenting competence and increased self-esteem from achieving home treatment has been
insufficiently investigated in the CF literature, given its importance for parenting. Recent studies by
McDonald and colleagues (2013) identified self-efficacy for home treatment as a dimension of
parenting a child with CF; Jessup and Parkinson (2010) reported parents’ fear of failing to administer
required treatment and Grossoehme and colleagues (2015) reported importance of parenting self-
efficacy for treatment adherence. My research identified how and why self-efficacy for CF home
treatment becomes inextricably linked to parenting competence, and discerned its effects on
parental identity and self-esteem. The current finding indicates that increasing parenting self-
efficacy for conducting home treatment could be a source of strength for parents to draw upon to
build confidence in establishing, managing and maintaining a home CF treatment regimen.
5.5.2.2 Gratitude, both for their child and for early surveillance.
Although sadness and fear are likely to always exist on some level or in some form, parents’
positive views of CF occurring on good days included a sense of gratitude for their child. Gratitude
existed in two forms. Firstly, parents appreciate their child is healthy relative to other children who
are inflicted with worse conditions. Secondly, parents are thankful for the unique opportunity to
track their child’s covert disease progression afforded to them by early surveillance. Each form of
gratitude will now be explained.
Gratitude for a child with CF through comparison has been interpreted as a result of
emotion-focussed coping, specifically the strategy of cognitive reappraisal. Cognitive reappraisal is
an emotion regulation strategy that functions to reduce negative emotion associated with a stressor,
whereby an individual reconceptualises a particular cognition so there is less burden associated with
136 Parenting Children with Cystic Fibrosis
it (Ray, McRae, Ochsner & Gross, 2010). In this instance, parents reconstruct their perceptions of
their child’s disease by comparing with children who have worse conditions.
This process has been labelled as social comparison (Festinger, 1964; Wills, 1981), and has
been shown to be a common coping strategy when people are confronted with health problems
(Van der Zee, Buunk, Sanderman, Botke & van den Bergh, 2000). For example, when individuals
faced with serious health issues make comparisons with selective information from their social
environment, it produces a favourable comparison situation that increases subjective well-being
(Christie & Barnard, 2012). Therefore, social comparison can serve as an adaptive process to a
threatening situation.
As parents in my research frequently visited their CF clinic, there was plenty of opportunity
for them to compare their child’s health with children who have worse health. A Victorian mother of
a 5 year old child said ‘I sort-of look around the hospital and think that we've got it pretty good
compared to a lot of people there so I try not to complain,’ and a Western Australian father of a 3
year old child said ‘yeah we kind-of think - at least we didn’t get what they’ve got, we got off easy.’
Comparing their family situation to families who experience worse conditions enabled parents to
positively reconstruct their perspective of their child’s illness within context of wider society.
Reconstructing perceptions of the child’s condition in this light resulted in the favourable affective
outcome of gratitude.
Research suggests that people who compare themselves favourably with others tend to
experience better health outcomes (Buunk, et al., 2012; Kwan, Love, Ryff & Essex, 2003). Even those
who receive information threatening to their self-concept, but who see themselves as better off than
the average person in their situation (known as downward comparison), tend to adapt more
successfully to failure or threat, and report greater life satisfaction and less psychological distress
(Kedia, Mussweiler & Linden, 2014; Stewart, Chipperfield, Ruthig & Heckhausen, 2013). Accordingly,
making downward social comparison has been shown to be self-protective by alleviating negative
emotional consequences of stressful encounters and threats to self-esteem (Bauer, Wrosch & Jobin,
2008; Heckhausen & Brim, 1997; Wills, 1981). Findings from my research suggest that social
comparisons allow parents of chronically ill children to self-evaluate in relation to others (Festinger,
1964; Kwan, et al., 2003; Moola, 2012; Tanner, 2007). Therefore, the current finding is consistent
with those of other studies that support the use of social comparison as a strategy to self-enhance
one’s own position and experiences.
The other reason parents experience gratitude, and the principal reason that parents enrol
their child in early surveillance is the opportunity to track their child’s disease progression. Parents
understand the unique opportunity afforded by early surveillance, thereby perceiving their situation
137 Parenting Children with Cystic Fibrosis
as lucky, resulting in feelings of gratitude (Cartwright, et al., 2011), as demonstrated by a Western
Australian mother of a 6 year old child:
I'm also grateful that we can go the hospital, I'm grateful for the bronchoscopy and the
CT. I'm just so grateful for what they're offering, how they do it, how they help her.
Everything about it really, I'm just so grateful. Something that is so unknown, you know
'oh we'll just give this a go, we don't really know if it's going to work.' That to me would
be so much more stressful. I'm just so appreciative of what's out there for CF.
Along with parents’ perception of the unique opportunity afforded by early
surveillance resulting in feelings of gratitude is the notion of perceived control and power over
disease progression. In this case, knowledge is power. These are the first findings, to my
knowledge, to describe this psychological phenomena for parents whose children undergo
early surveillance for covert disease progression, and are important to communicate to other
paediatric CF centres adopting the early surveillance framework (e.g., Utrecht University in The
Netherlands for example).
5.5.2.3 Perceived control and power through certainty of disease severity from early
surveillance results.
The primary objective of early surveillance is to detect disease for the purpose of
administering proactive intervention. For parents, the primary objective of disease detection and
successive intervention neutralises their primary concern of CF: that is, uncertainty in morbidity and
life expectancy. To explain how parents feel a sense of control and power over their child’s disease
progression is to reveal how early surveillance outcomes respond to parents’ perceptions of CF. To
explain further, all-encompassing uncertainty becomes the lived experience for parents of children
with CF, but becomes neutralised by assurance from perceived control. Power from perceived
control is a reaction to assurance generated from receiving detailed information about a child’s
covert disease progression and subsequent early intervention.
Assurance of child health as a result of this information may be a unique construction for
parents whose children undergo early surveillance. The following quote from a Western Australian
mother whose child was 3 years old demonstrates how she felt assured her child was being treated
as early as possible with the most sensitive measures available, ‘I think it's nice to have that
reassurance that everything that you're going through every day and the disruption that it causes to
our family life is actually making a difference…it's nice to know that all of that is having a bearing on
her health.’
Knowledge of covert disease progression and assurance that early intervention is
administered in a proactive manner creates a sense of control for parents, which gives them a sense
of power. The following quote from a Western Australian mother of a 12 month old baby
138 Parenting Children with Cystic Fibrosis
demonstrates that parents feel a sense of control over their child’s disease progression because of
the assurance given by early surveillance that their child is being treated as early as possible with the
most sensitive measures available:
it’s reassuring because we’re so new to what CF is, and things like that. You know, you’re
not sure, has she already got something? So yeah, that’s what I’m looking forward to for
the next one. You know, to know that she’s ok. You know, using it in a positive way so
that if she’s not well then, bang they can get on top of it. I feel like I have some control
over something that I can’t see. So I think it’s great, I think it’s better than not knowing.
Rodriquez and colleagues (2012) suggested that the most stressful aspect of caring for a
child with a chronic condition may be a lack of control, that is, parents may feel a loss of control
when, for example, they are uncertain about their child’s future prognosis. The quotes following the
diagram demonstrate that parents felt a sense of control and power over disease progression
through knowledge provided by early surveillance. The diagram below depicts how knowledge of
covert disease progression from early surveillance and assurance of early treatment generates a
sense of control over disease progression.
Figure 8. Relationships between knowledge, assurance and control
Victorian mother, 3 year old child - I think having more information gives me control, I
like control…my goal is to be in control and information helps me to get that control.
Victorian father, 5 year old child - I can go 'ok, that's done and this is what it means,' and
I can move on. I just like to know, it's a bit of control.
Victorian mother, 4 year old child - it's important to know. Knowledge is power and
that's why we're doing this, we want to know.
Western Australian mother, 6 year old child - we still had a better understanding of her
X-rays, so the more knowledge you have the better you feel…yeah, knowledge is power.
139 Parenting Children with Cystic Fibrosis
Therefore, on days when parents have a positive outlook on their child’s life, and on CF in
general, knowing they are recipients of the most detailed information about CF disease progression
that is available for their child gives parents a sense of control over disease progression. Perceived
power and control over disease progression are a direct consequence of early surveillance, and
become of central importance to parents on a long-term basis, thereby functioning as a lived
experience for parents whose children undergo early surveillance of CF lung disease.
On a final note, it is worth highlighting that whilst parents in my research felt power and
control over their child’s disease progression on their good days, they did not appear to experience
helplessness on their bad days. Parents’ feelings of helplessness as a result of their child’s CF
(Carpenter & Narsavage, 2004; Jessup & Parkinson, 2010) and other progressive chronic diagnoses
has been supported in the literature (Hechlerl, et al., 2011; Pelentsov, Laws & Esterman, 2015), but
was not a finding in my research (however feelings of helplessness as a result of early surveillance
procedures will be described in a later theme). That parents in my research were not found to
experience helplessness may be due to their perceptions of control and power over disease
progression afforded by early surveillance neutralising the feelings of helplessness that have been
reported in other studies with parents of children with CF (Coyne, 1997; Goldbeck, et al., 2010;
Staab, et al., 1998).
5.5.3 Fluctuation between good days and bad days.
We now know what parents experience on particular days, but how do those days fit together
to create a more global view of parents’ lived experience? Parents expressed how they shifted
between a positive and a negative perspective on CF. Parents appeared to fluctuate between good
days and bad, those positive and negative psychological constructs. A continual cycle between good
days and bad days; fear and chronic sorrow fluctuate between parental competence and satisfaction,
gratitude and perceived control. Duff and Oxley (2007) described this process as oscillating phases of
chronic sorrow with phases of normality and routine, punctuated with intense sadness and
mourning.
A Victorian couple whose child was 2 years of age described how they fluctuated between
good days and bad days when the mother stated, ‘that's our life now, but you don’t always feel this
level-headed, sometimes you have your down times and you think 'why did this have to happen,' but
it's just the way it is, that's what we've been dealt and you just have to deal with it.’ The father
responded to the mother’s statement by adding, ‘I just stress about the condition in general. Some
days you stress about it more than others. Some days are better than others.’ Others have also
reported a continuously fluctuating cycle for parents of children with CF between distress and
normalcy, adversity and adjustment (Duff & Brownlee, 2008; Glasscoe & Smith, 2008, 2011; Jessup &
Parkinson, 2010).
140 Parenting Children with Cystic Fibrosis
It was not determined how often, or why, parents fluctuate between these two perspectives,
for how long they persist with either of those outlooks, or reasons why a particular psychological
state comes about. However, the substance of each of those types of days and psychological states is
important information for both clinicians and other mental health professionals working with these
families. With this information, HCPs can better understand parents’ experiences of their child’s CF
that can help with clinician-parent interactions, implicating rapport and trust development.
In summary, a CF diagnosis brings much psychological distress for parents, and may continue
beyond the diagnosis phase. Fear is induced by uncertainty in morbidity and mortality and chronic
sorrow is induced by loss of child, parent and family expectations. Parents managed their fear
through a number of mechanisms including social comparison and social support. Moreover, a
number of parental characteristics were identified that may contribute to coping with fear associated
with paediatric CF, for example, parents of children 3 years or older. Parents’ lived experience
includes a fluctuation between good days and bad days, a cyclical journey between the former and
the latter (Gjengedal, et al., 2003). The good days are filled with feelings of high parental self-esteem,
gratitude and perceived control and power over disease progression.
CF treatment management becomes a major component of family life and parent identity, and
therefore merges into new normal; the redefined sense of self and family life. Psychologically,
parents thrive on achieving the demanding home treatment regimen because by maintaining
treatment management, perceived parenting competence supports the re-imagined parent identity
as medical carer, potentially facilitating parental self-esteem. Moreover, perceived control over
disease progression also becomes part of the new normal for parents in my research, which
generates a sense of power from knowledge and gratitude from comparisons. Therefore, amongst
the fear and chronic sorrow, parents experience competence and self-esteem, gratitude, control and
power.
5.6 Early surveillance is a significant event
To this point, findings have described parents’ lived experience of CF; the diagnosis, the
unknown that, with time and experience, turns into the understood and eventually accepted. The
initial experience of CF includes introduction, learning and assimilation of CF and its treatment
management into family life. How introduction of CF affects parents’ well-being more generally has
also been outlined, with demonstration of how parents fluctuate between good days and bad days,
of what each of those types of days consist, and how parents are affected by them. Introduction to,
and learning about, early surveillance for CF lung disease has also been discussed. This theme shifts
focus from how CF and early surveillance come to be understood, to demonstrate how early
surveillance is constructed by parents as a significant event: the meanings attributed to it for
perceptions of themselves as parents including factors that contribute to the process, and how early
141 Parenting Children with Cystic Fibrosis
surveillance can affect family life. This theme is about the experience of early surveillance, whereas
prior discussion was about how early surveillance comes to be understood. Therefore, this theme
describes parents’ lived experience of early surveillance for CF lung disease.
To explore the complexity of this theme, three sub-themes were identified that detail the
above varied aspects involved with early surveillance for parents in my research. The sub-themes
were: indicator of treatment efforts and examination of parenting competence; parental outcomes
directly from early surveillance; and fear from diagnosis exacerbated each annual review can result in
anxiety.
5.6.1 Indicator of treatment efforts and an examination of parenting competency.
A common perception held by parents and society more generally is that children, and their
development, outcomes and achievements are a reflection of their parents’ competency to raise and
care for them. The link between child outcomes and perceived parenting competency extends to
early surveillance; sensitive detection of early disease progression is scrutinised against parents’
treatment efforts and health awareness throughout the year. As a Western Australian father of a 5
year old child stated, ‘all year you try to make sure you’re doing the best thing for him so it’s always
almost like an exam for us. It feels like an exam to us so you sort-of just really want to know whether
you’ve passed and whether he’s passed.’
Parents whose children received favourable annual review results expressed belief that their
treatment efforts had a beneficial effect on their child’s health, which created a sense of parenting
competence. An excerpt from a Western Australian mother of a 5 year old child highlights
accountability she felt for her child’s favourable annual review results, ’ it is like you are studying for
a test to a certain extent and have you done well. You feel fantastic when you get a great result, you
feel really good about your parenting.’
Inversely, negative annual review results are constructed by parents as a reflection of their
inability to conduct adequate levels of home CF treatment. This concerning finding of accountability
for unfavourable annual review results is demonstrated by a Victorian mother whose child was 4
years old, she stated, ‘you know when we get a bad result, you second-guess yourself. Could I have
done something differently? Where did he pick it up? Was it our fault he got it? How can I be a better
parent?’ Bluebond-Langner (1996) found that parents’ sense of competence was threatened when a
child was admitted to hospital for an exacerbation. Collectively, these results indicate that parents’
perceived competence in relation to their child’s health can be adversely affected by events attesting
to a child’s early disease progression.
Self-directed blame for unfavourable annual review results appears to be a consequential
interaction of the model of home CF treatment and the sensitivity of measuring covert disease
progression because of the link parents make to their child’s home treatment. The inextricable links
142 Parenting Children with Cystic Fibrosis
between home treatment and sensitive measures of disease progression may mean that blame is
unavoidable for parents whose children undergo early surveillance. Given the risk for depression,
post-traumatic stress syndrome (PTSS) and anxiety from parental self-blame for paediatric chronic
illness (Greening & Stoppelbein, 2007), such information could educate HCPs about parents’
constructions of early surveillance outcomes and inform clinical care pathways that might target
adverse parental outcomes associated with self-blame for diagnosis and/or negative test results.
High self-efficacy for treatment management resulted in parents believing that their treatment
efforts will have an effect on their child’s health, hence attributions of internal locus of control for
child health outcomes. Parenting self-efficacy has been explained elsewhere, and locus of control
refers to the extent to which individuals believe they can control events affecting them (Rotter,
1966). An internal locus of control orientation is a belief about whether the outcomes of actions are
contingent on what a person does, whereas an external locus of control orientation is a belief that
events are outside of personal control (Zimbardo, 1985). Extensive literature across a multitude of
disciplines attests to the relationship between high self-efficacy and internal locus of control,
including vocational and health behaviour (Joo, Lim & Kim, 2013; Roddenberry & Renk, 2010;
Strauser, Ketz & Keim, 2002).
The finding of attributions of internal locus of control for child health does not support
previous research on parents’ constructions of their role in their child’s health, with a number of
studies showing that parents exhibited an external locus of control for their child’s health. Parents
believed factors such as fate, religion or medical intervention were the primary causes of child
health (Lawoko & Soares 2002; Menahem, et al., 2008). The reason for this inconsistency is not clear
but it may be due to the level of home treatment required by parents of children with CF. For
example, parents of children with chronic kidney disease who were required to administer home
treatment including dialysis and injections felt at fault if their child developed infections or other
complications (Tong, et al., 2010). Attribution of internal locus of control for health outcomes and
high-self-efficacy for treatment maintenance in terms of parents’ constructions of their child’s health
and disease progression is an important finding in my research. It illuminates how parents interpret
information from early surveillance, what meanings they attribute to it, and informs what effect it
has on them. A passage from a Victorian father whose child was 4 years old illustrates how he felt
when his child’s annual review results were positive. He stated, ‘I guess it's a confidence thing too. If
they say that her lungs are clear then it makes you feel good as a parent.’ On the contrary, a
Victorian mother of a 12 month old baby expressed how she felt when her child’s results were
negative. She said, ‘she had quite a lot of damage in one part of her lung so that was a bit
confronting for us that day. It felt like we had done something wrong as parents, even though we
fully understand that not to be the case, you can't help how you feel no matter what you think, kind-
143 Parenting Children with Cystic Fibrosis
of thing.’ The diagram on the following page depicts how early surveillance results are constructed
within the parental role, and how they are attributed meanings of parenting competence, which
may affect parental identity and parental self-esteem. Note the contribution of early surveillance to
the effects of parenting self-efficacy on parenting competence beyond the diagram outlined on page
132 showing a link between treatment management and parenting competence.
Figure 9. How early surveillance influences perceived parenting competence and possibly self-
esteem
The current findings go beyond existing knowledge by highlighting how high levels of
parental involvement in CF care, high importance attributed to the care and perceived accountability
of child health all contribute to what it means to be a parent of a child with CF. Moreover, the
current findings add to knowledge by identifying how early surveillance results may disrupt the sense
of mastery parents feel about their child’s treatment, which can influence perceptions of themselves
as parents.
144 Parenting Children with Cystic Fibrosis
Finally, an external health locus of control for parents of chronically ill children has been
shown to predict parental anxiety (Scrimin, Haynes, Altoè, Bornstein & Axia, 2009) and also has been
associated with parenting stress (Hassall, Rose & McDonald, 2005). Green (2004) showed that
mothers high on external locus of control felt more subjective caregiver burden than mothers who
were high on internal locus of control. Moreover, studies examining personality attributes and well-
being have reported that internal locus of control and high self-efficacy is associated with better
psychological well-being (DeNeve & Cooper, 1998; Roddenberry & Renk, 2010; Streisand, Swift,
Wickmark, Chen & Holmes, 2005). How early surveillance affects parents’ psychological well-being
warrants further investigation because it is plausible that such interactions exist between the early
surveillance experience and parental psychological well-being. For example, attributions of internal
locus of control and high self-efficacy may function as buffers against stress associated with receiving
negative early surveillance results, and at the same time diminish self-esteem that could exacerbate
stress (Abouserie, 1994). In summary, introduction of CF into the family system requires
implementation of a new set of parenting responsibilities as part of a redefined and medicalised
parental role. Parents whose children undergo early surveillance construct the sensitive measures of
disease progression as an indicator – a judgement – of their ability to achieve these new parenting
responsibilities and maintain child health. Therefore, this sub-theme demonstrated how early
surveillance is constructed as an indicator of treatment efforts and competency throughout the year
and as an examination of parenting ability, and if identified meanings attributed to those
constructions affected parental identity and parental self-esteem.
5.6.2 Parental outcomes directly from early surveillance.
Significance attributed to the early surveillance event by parents meant that a number of
psychological phenomena where experienced at the time of annual review. Parents experienced
feelings of lack of control and helplessness when relinquishing their child for the procedures of early
surveillance, that is, for the bronchoscopy and CT scan under general anaesthetic (GA). Parents also
felt a sense of altruism for future CF generations as part of their decision to involve their child in early
surveillance. Therefore, parents could simultaneously experience positive and negative outcomes on
the day of their child’s annual review.
5.6.2.1 Lack of control and helplessness at time of relinquishment for medical procedures.
Relinquishing a child to both actual harm (i.e., the procedures) and then further potential
harm and threat (i.e., complications during procedures) is the most confronting component of early
surveillance for parents - psychologically and behaviourally. Relinquishment of a child, the heart of
the parents’ life world, into a threatening situation they do not control causes feelings of
helplessness. For parents of younger children, they are often relinquishing their children to people
they have never met before.
145 Parenting Children with Cystic Fibrosis
Each year parents are required to relinquish their child for early surveillance procedures, and
each year parents generally feel a sense of helplessness about how their child will react to the GA, as
well as how they will fare during the medical procedures. A Western Australian mother of a 6 year
old child expressed her feelings of helplessness when she said, ‘it's very difficult because that part is
out of your control and because then you walk away and you don't have sight of your child and that's
extremely heart-wrenching because you have lost control and that loss of control…in your child's life
when they need you the most.’ A Victorian father of a 4 year old child echoed this sentiment when
he stated, ‘just to know that something could potentially go wrong or you start thinking 'what if she
never wakes up,' and those sorts of things… it can be, the moment that you're in the realisation of
what you're doing or what the risks are, sometimes become rife and obvious. Sometimes my mind
can start to get away from me and you just get to a point where you feel so helpless.’ The current
finding is consistent with those of other studies (Bull & Grogan, 2010; Hughes & Callery, 2004;
Salgado, et al., 2011; Vessey, Bogetz, Caserza, Liu & Cassidy, 1994) that show relinquishing children
for medical procedures is generally a confronting and uncomfortable experience for parents.
The psychological consequences of relinquishment (i.e., lack of control and helplessness)
generally become more manageable over time as expectations are developed based on previous
experience. It is therefore assumed that as parents encounter these medical procedures more, most
will cope better over time (Utens, et al., 2000). As relinquishment of their child is always a
characteristic of early surveillance, as is uncertainty in child safety, so is this sense of helplessness
during this time. Whilst lack of control and feelings of helplessness become easier to cope with over
time and with experience, they never completely disappear.
In accordance with the finding identified in sub-theme understanding early surveillance - that
over time, parents experience transition from no expectations to some expectations of the program,
the current finding suggests that time and experience with early surveillance may facilitate parents’
ability to manage and cope better with relinquishing their child for medical procedures, as well as
coping with their own thoughts and emotions associated with relinquishment. The following excerpts
demonstrate how expectations helped parents of children aged more than 3 years to cope with the
medical procedures, whereas parents of younger children did not express this particular coping
mechanism. Parents of older children expressed the evolution of their coping process on which
younger parents did not comment:
Western Australian mother, 4 year old child - I would say that it's easier to handle. It all
just comes back to experience and understanding again, knowing what's in store for you.
Knowing how the procedures are going to go…definitely things have gotten better over
time of knowing what to do and how to do it. It all just comes down to experience.
146 Parenting Children with Cystic Fibrosis
Western Australian father, 5 year old child - Yeah, it’s probably got a bit easier I suppose.
The first ones, you know, when he was a baby, were sort-of, you know, a little bit
harder…it’s probably just got a little bit easier as the years have gone on…so yeah, the
way we deal with it. We know a lot more about it now. We sort-of know how to cope
with it.
Literature from diverse disciplines (e.g., medicine, technology and psychology) across various
situations (Borup, Brown, Konrad & Van Lente, 2006; Faasse & Petrie, 2013; Hauck, Fenwick, Downie
& Butt, 2007; Liao, Gramann, Feng, DeáK & Li, 2011) shows how expectations for an event help
people to manage similar future events. Expectations provide a framework through which
psychological boundaries created during early experiences control those psychological processes and
outcomes for future experiences of similar events. In this way, and similar to creating expectations
about the family and child after diagnosis, parents can reduce initially boundless thoughts and ideas
of relinquishing children for procedures (and of the early surveillance event more broadly), which in
turn helps parent to cope by reducing stress associated with those earlier boundless ideas and
thoughts. Therefore, interventions aimed at supporting parents through their child’s early
surveillance procedures could encompass the need to frame expectations.
5.6.2.2 Altruism for future CF generations.
The opportunity to participate in early surveillance with the intention of contributing to
future CF generations’ lives through research is conceptualised in my research as altruism. As a result
of their child’s early surveillance involvement, parents described a sense of altruism. Parents who
asserted altruism as their primary reason for early surveillance participation attributed high
importance to research participation. Therefore, altruism in this instance is interpreted both as a
consideration for, and as a beneficial outcome of, research participation. Parents’ altruistic
disposition is demonstrated by A Western Australian mother whose child was 5 years old, ‘I see study
participation as quite altruistic because any kind-of benefit is not necessarily going to rain down on
XXX…in my eyes, we had to be altruistic because the benefits aren't going to rain down on your child
but for other families, it might.’ This point of view was reflected by a Victorian father of a 2 year old
child, ‘the whole idea that it's going to go to contributing to people in terms of future benefits is a
driving force for me.’
Perceived direct clinical benefit for their child was another reason for participation. For these
parents, altruism appeared as more of a bonus beneficial outcome than as a primary reason for
research participation. These parents placed more importance on their child’s direct clinical benefit
than research for medical advancement, but still held importance for research participation.
Cartwright, et al. (2011) found the same dominant two reasons for parents’ consenting their children
to a randomised controlled trial. A quote from a Western Australian mother of a 4 year old child
147 Parenting Children with Cystic Fibrosis
indicates her reasons for agreeing to early surveillance for her child, ‘I hope it will help my kids
hopefully more than anything, but also it’s going to help future generations…I'm grateful that it's
there, I'm grateful that I can participate in it. Firstly to get information for ourselves but secondly to
help with research as well.’
All parents placed some level of importance on being altruistic for research and scientific
advancement purposes. They all acknowledged that early surveillance is something they wished their
child did not have to do, but they certify its importance for their child’s health and for future CF
generations. The primary motivation behind altruism for future CF generations was that because
children today have been afforded a longer and improved quality of life by parents of previous CF
generations subscribing their children to such research programs, that parents wanted other children
to experience the same benefit. A Western Australian mother of a 12 month old baby spoke about
why she felt importance of participating in early surveillance, ‘ I feel like it's something little that we
can do to make a difference for the kids in the future. I mean really, so much progress has been made
and I feel like that's because the parents before us have sacrificed so much and gone through so many
trials so I’d like to think that we can do that for parents coming through…there are families who have
gone before them who have done the research and that's why our kids are doing so well now.’
However, an alternative explanation for this sense of altruism is that parents felt a sense of
obligation or duty to continue the trend towards research participation, with less regard for their
own innate thoughts and feelings about research participation.
From the point of damage to parental identity is the notion that an altruistic tendency may
aid in its repair, and therefore contribute to effective parental coping. Feelings of altruism may form
a counter-perspective of parental identity, damaged by diagnosis or receipt of unfavourable review
results. Therefore, parents may draw on what positive aspects of the program they can to cope with
the negative ones. To that end, negative aspects of the program (i.e., blame and guilt for negative
results and lack of control and helpless due to relinquishment of a child for medical procedures) are
intense and acute (i.e., experienced around the time of annual review), whereas positive aspects
(i.e., perceived control and power over disease progression, parental competence and satisfaction
and altruism) exist on a long-term scale. To this extent, it appears that perceived long-term
outcomes aid parental coping with the intense, acute experiences around the time of annual review.
It may also be that altruism experienced acutely around the time of annual review is associated with
gratitude experienced on a longer-term basis on those days where parents have a positive outlook
on their child’s life. This idea, too, points to a relationship between acute experiences at the time of
annual review for longer-term outcomes.
Literature exists on parents’ initial decisions about involving their child in medical research,
mostly in oncology (e.g., Chappuy, et al., 2006) and neonatology research (e.g., Jansen-van der
148 Parenting Children with Cystic Fibrosis
Weide). Altruistic motivation has been reported as a leading justification for parental consent in
paediatric medical research, from polio vaccines in the 1950’s (Clausen, Seidenfeld, Deasy & Clausen,
1954) and vaccine trials in the 1990s (Langley, Halperin, Mills & Eastwood, 1998), to more recent
vaccine trials (Chantler, et al., 2007). The current finding is consistent with those of other studies
(e.g., Chanter, et al., 2007; Chudleigh, et al., 2013; Steele, et al., 2014) and suggests that parents
want to advance medical science, but primarily they want to help other children (and families) in the
same situation as themselves. It is possible these results are due to participation viewed as a social
obligation, particularly if their child benefited from prior medicine advancement that was a result of
research participation by families previously in their situation.
5.6.3 Fear from diagnosis exacerbated at annual review.
Fear experienced around the time of annual review is distinguished from lack of control and
helplessness as direct outcomes of early surveillance because fear originates at diagnosis, continues
as part of the lived experience of parenting a child with CF, and is then exacerbated at annual review.
It is perceived seriousness and risk of medical procedures that assigns additional reasons to
previously experienced fear. The processes leading to fear differ from processes leading to lack of
control and helplessness, in that medical procedures serve to increase, or intensify, fear experienced
more globally, which was expressed by parents as occurring on their bad days.
The GA and the medical procedures were the components of the early surveillance program
that concerned parents the most (Chudleigh, et al., 2013). Accordingly, the GA and medical
procedures engender additional aspects to fear associated with CF. A Western Australian mother of a
2 year old child said, ‘I’m scared of the GA more than anything else.’ Additional dimensions of fear
were perceived risk of the medical procedures, potential trauma for the child, as well as lack of
control and helplessness due to relinquishment. Due to the nature of early surveillance for CF lung
disease, these additional reasons for fear have been reported for parents of children undergoing
surgery or requiring hospital admissions (Bull & Grogan, 2010; Himes, Munyer & Henly 2003;
Menahem, et al., 2008; Utens, et al., 2000).
Fear described in interviews appeared solitary and personal, that was often expressed to a
partner or family member and generally not expressed within the family domain. Parents did
however report being withdrawn from their family around the time of annual review. For example, a
Victorian father of a 5 year old child stated, ‘I just keep it to myself. When I stress, I try not to show it
too much. I'm probably more reserved around the kids during that time.’ Similarly, a Victorian mother
of a 2 year old child expressed her withdrawing behaviours in the time leading up to her child’s
annual review, ‘I get a bit apprehensive around that time…it probably brings my mood down a bit, it
makes me feel a bit deflated and I guess a little bit less engaged with the family for that period of
149 Parenting Children with Cystic Fibrosis
time.’ Though a limited number of parents recalled withdrawing from their families, and only for
relatively short periods of time prior to their child’s annual review, behavioural disengagement by
parents of children with CF has been associated with child and parent maladjustment (Wong &
Heriot, 2008), and should therefore be of concern to HCPs and researchers.
Parents’ primary coping mechanism for fear and in the lead-up to the annual review was
social support from friends, family and/or a partner. An example of how parents drew on personal
support in the lead-up to annual review came from a Western Australian mother of a 2 year old child
who said, ‘well we talk about it, well (laughs) I'll talk to XXX (husband) about it, and I've talked to my
friends about it. You know, I will talk about it.’ The current finding is consistent with CF research and
other research showing that parents whose children undergo surgery commonly use social support
as a primary coping mechanism (Grossoehme, et al., 2014; Hughes & Callery, 2004; Salgado, et al.,
2011).
On the day of the annual review, the dominant coping strategy for relinquishing children for
medical procedures was the distraction technique, an emotion-focussed coping strategy (Lazarus &
Folkman, 1984) functioning to divert attention away from the stress of relinquishment until such
time that the child was back with the parent. Distraction techniques included talking on a mobile
phone or with the person who accompanied them, reading a magazine or visiting the café or gift
shop. Examples from parents using distraction to cope include a Western Australian father of 4 year
old child who said, ‘we usually grab a coffee and walk around and buy a paper, go for a walk. You
know, try and do tasks and things, play on my phone,’ and a Western Australian mother of a 5 year
old child said, ‘I usually take that time to run errands around the hospital like go and pick up some
medications from pharmacy so that we're not waiting around …so that certainly takes your mind off
it…for me, if I'm not thinking about it at the time, not that it's not happening, but I can deal with it
better.’
The excerpts are from parents whose children are over 3 years of age, suggesting that
coping with early surveillance may be a function of time and experience to learn effective coping
techniques. Therefore, further understanding of parental factors that characterise parents who
experience increased fear at the time of medical procedures, and how they cope will inform if, and
what, interventions may help to alleviate fear during relinquishment of children for medical
procedures.
Behavioural disengagement and distraction are considered maladaptive coping techniques
(Carver, et al., 1989). They have been shown to reduce emotional and social quality of life (Kertz,
Stevens, McHugh & Björgvinsson, 2015; Staab; et al., 1998) and increase depression, anxiety and
stress (Sheehan, et al., 2014) both in parents of children with CF and adults with CF. Though parents
described using these coping strategies for brief periods of time, due to the cross-sectional nature of
150 Parenting Children with Cystic Fibrosis
my research, I was not able to discern their temporal nature. Given my research findings, AREST CF
clinicians and researchers should be cognisant of when parents need to cope with early surveillance,
and that this period of time extends from the days prior to annual review, including the day of
annual review and then ensues until receipt of early surveillance results. A Victorian mother of a 4
year old child explained this concept comprehensively when she stated what she needed to manage
and when, and how she reframed the situation as leading to a perceived positive outcome:
I think at least the 48 hours before, and even the week before, we give XXX (child with
CF) a present to take the hospital to have for when he wakes up after the anaesthetic,
just to try and make the process a bit easier. So yeah, there’s a bit of of dread…so I’m
trying to, I think it’s a really beneficial program so, but there’s also the worry of what
results we’re going to get. So you kind-of think about this all before you go, and then
it’s about actually getting there. You know, the drive there, parking. Parking drives us
up the wall. So yeah, I think there’s never like, we kind-of just go ‘well, we know that
shit day is coming up.’ You don’t look forward to it obviously, I just look forward to
knowing how the kids are progressing. That’s probably the only positive thing as a
parent.’
Currently there is only anecdotal evidence and clinicians’ assumptions about points in the
early surveillance process with which parents may be struggling. Beyond simple awareness of when
and how parents cope, a support program or intervention strategy could be developed and
implemented that is responsive to the findings in my research about parental coping strategies and
techniques. Finally, if chronic sorrow was indeed experienced as a result of CF diagnosis, it too, may
be exacerbated by CF coming to the forefront of parents’ minds as the significant event of early
surveillance looms. Perceived significance of the event may exacerbate chronic sorrow around the
time of annual review as a reminder of the diagnosis and prognosis of CF itself.
Chronic sorrow theory suggests that an exacerbation can occur as a result of a medical crisis
or event that magnifies the loss and disparity between reality and the life once imagined (Eakes, et
al., 1998). The event can therefore trigger a return of profound sadness. Parents expressed
experiences of recurring sadness throughout the year (as discussed in the theme good days and bad;
fluctuation between positive and negative), with the annual review emerging as a trigger that
exacerbates chronic sorrow. Bluebond-Langner (1996) described the content and process of triggers
as information moving back and forth, that is, devastating characteristics of CF (namely morbidity
and mortality) come to the forefront of parents’ minds where it has been pushed to the back of the
mind for the most part, resulting in those emotional and cognitive responses experienced at
diagnosis “by thinking about it all over again” (Bluebond-Langner, 996, pp. 150)
151 Parenting Children with Cystic Fibrosis
Reports in the literature portray events associated with a child’s CF as intruders to a
generally dormant condition in the early years of life, which can make their presence known to
parents and interrupt the normality that has been redefined for the family (Bluebond-Langner, 1996;
Glasscoe & Smith, 2011). It appears that for parents in my research, the early introduction of a
significant event (that is, early surveillance) becomes part of the redefined sense of what is normal
for the family. Not to minimise the effect this has on parents, but they appear to assimilate this event
(and its associated information about their child’s disease progression) into their redefined lives
where family life appears to remain stable throughout (and as a result of) the event. However, it
remains that fear and potentially chronic sorrow can be re-visited, and warrants further
investigation.
Finally, a pertinent consideration must be that, although logically (and described in the
current finding) parents cope better with feelings of lack of control and helplessness, and fear over
time, it is possible parents experience PTSS or clinical levels of depression associated with their
child’s ongoing surgical procedures and/or subsequent treatment. For example, parental depression
can be prevalent up to 1 month after a child’s surgery (Ben-Amity, et al., 2006) but generally not 12
months later (Menahem, et al., 2008). The current finding and other literature (Bearden, Feinstein &
Cohen, 2012; Fincher, Shaw & Ramelet, 2012: Jay & Elliot, 1990) highlights potential need for short-
term intervention that supports parents’ well-being prior to, and following their child’s early
surveillance procedures.
Whilst parents generally appear resilient to the burden of early surveillance, the results do
need to be interpreted with caution. Firstly, there is the possibly that PTSS or clinical levels of
depression and/or anxiety are experienced by parents who chose not to be part of the research.
Secondly, as this research is based on retrospective data, parents may indeed experience withdrawal
and/or PTSS symptoms annually around the time of their child’s early surveillance procedures but
that were not recalled during interviews.
In summary, it appears that perceived accountability for child health may be a unique
psychological phenomenon for parents in my research. The accountability is constructed from the
nature of CF management and its association with parents’ constructions of detailed information
about a child’s health. The consequence of this association is that parents use their child’s early
surveillance results as an indicator of their treatment efforts, which influences perceived parenting
competency. Parents’ experience of their child’s early surveillance included feelings of lack of control
and helplessness from relinquishing their child for medical procedures, however parents tend to
manage these feelings better over time. Parents also experienced altruism as a positive consequence
of participating in medical research. Fear experienced at diagnosis can be exacerbated at the annual
review due to the risk in medical procedures, potential trauma to the child and parents’ feelings of
152 Parenting Children with Cystic Fibrosis
helplessness. By virtue of annual review results being unique to parents in my research, the
experience of early surveillance and its outcomes produce unique parental psychological outcomes
that are independent of CF.
Accordingly, the current theme demonstrated that early surveillance is constructed as a
significant event that has boundaries and evokes emotion. Boundaries of early surveillance
circumvent the reminder of impending annual review to receipt, and any consequences, of review
results. The significance of the event appears to bring CF to the forefront of family focus. It is not
that parents ever forget their child has a life-threatening condition, but potential adverse
consequences of the impending annual review can aggravate psychological aspects of CF that have
been somewhat supressed or negated throughout the year. Aggravation causes exacerbation of
those psychological parental outcomes of having a child with CF.
Thus far, the current theme (and its sub-themes) has begun to describe parents’ lived
experience of CF within the context of early surveillance and how there are confronting, and
sometimes conflicting, psychological processes occurring. Moreover, both beneficial and adverse
outcomes for parents of children with CF within context of early surveillance have thus far been
described. Conflicting responses caused between presence of both positive and negative outcomes is
the matter of the next theme early surveillance is in the best interest of my child’s health.
5.7 Early surveillance is in the best interest of my child
This theme continues the presentation of parents’ experiences and constructions of their
child’s early surveillance, and articulates parents’ ultimate perception that early surveillance will
benefit their child’s long-term health because it may save, or at least extend, their child’s life. The
purpose of this theme is to describe the primary reason for parents’ decision to enrol their child in
early surveillance, concluding with discussion about what potential effects the decision to participate
in early surveillance has on parents. Sub-themes identified to illustrate these constructed thoughts
and experiences include; perceived clinical benefit to child health – presence of the therapeutic
misconception?, and cognitive dissonance results in anxiety and/or ambivalence.
5.7.1 Perceived clinical benefit to child health – presence of the therapeutic
misconception?
The primary clinical aim of early surveillance is to detect disease early so that therapeutic
treatments can be introduced, with the intention to try and prevent disease progression, or at least
delay it. The primary research aim is to understand the pathogenesis of lung disease in early life and
identify modalities that allow clinicians to detect lung disease in its early stages. The primary aim for
parents is to preserve their child’s life through ensuring any disease progression is treated as early as
possible. Parents’ perceptions that early surveillance will directly benefit their child through
improved clinical health outcomes is illustrated by quotes from a Western Australian mother of a 5
153 Parenting Children with Cystic Fibrosis
year old child who said, ‘it’s one of those things we know we have to do because of the benefits of it,
so there’s that mixed with the feelings that you don’t want to do it…we prioritise what she needs as
opposed to our stress,’ and from a Western Australian father of an 18 month old child who said, ‘it’s
got to be worth it because you can give your kids a few extra years… it’s the fact that it can give your
child however many years of life and good quality of life.’
Belief in improved child health outcomes is an artefact of parents’ constructions that early
surveillance will result in direct clinical benefit to the child. However, the clinical benefits ascribed to
early disease surveillance are based on logical inference rather than evidence-based clinical
outcomes. That is, that by detecting disease early in life, treatment may be instigated early that
could delay or slow down disease progression. Surveillance of CF lung disease itself is not a
therapeutic intervention. In absence of evidence-based outcomes, there are a number of reasons for
this circumstance. As CF treatment and standards of care have changed dramatically over recent
years it is difficult, if not impossible, to attribute clinical health improvements to the early
surveillance processes. Also, as early surveillance is still relatively in its infancy, long-term health
outcomes are yet to be characterised.
However, there are a number of indications that early surveillance leads to better health
outcomes for children with CF. Evidence to date suggesting intervention as a result of early
surveillance modifies clinical health outcomes is provided through comparison of early surveillance
data with Australian CF registry data. This comparison shows that children with CF under 7 years of
age in Western Australia and Victoria are healthier than children in other states, indicated by
nutritional indices and pulmonary function (Cystic Fibrosis Australia, 2013). The issue for valid
comparison is lack of control data for confounding factors that may influence these data, for
example, environmental factors associated with socio-economic status may affect a child’s
respiratory health (Neidell, 2004; Thakur, et al., 2013). Another source of evidence suggesting early
intervention leads to improved health outcomes can be inferred from NBS research, which shows
that early diagnosis and subsequent early introduction of CF treatment benefits children’s nutritional
outcomes (Farrell, et al. 2001).
Without evidence-based information of clinical benefit, and inferences made from CF registry
data and NBS data, does this mean the therapeutic misconception is present for parents who believe
their child will benefit clinically from early surveillance of CF lung disease? Consideration of the term
will enlighten the reader to its conception as well as its potential existence in the population under
investigation. Therapeutic misconception is a common ethical problem encountered in human
participants research. First described by Appelbaum in 1982, many people were unaware of the
differences within a clinical setting between research participation and receiving treatment. Instead
154 Parenting Children with Cystic Fibrosis
the belief was that therapy and research were governed by the same goal, that is, to advance the
individual patient’s best interest.
Although it may be a consequence of research participation, the primary goal of medical
research is to generate data that could lead to improved care for future patients (Dresser, 2002). The
therapeutic misconception describes the belief that the purpose of a clinical trial is to directly
benefit the individual patient rather than to gather data to develop scientific knowledge (National
Bioethics Advisory Commission). Put simply, it is the belief of patients that by entering a research
study, they will benefit directly from participating, regardless of whether they actually will or not.
Although the therapeutic misconception has been well described (Joffe & Miller, 2006), it has
not been considered in the context where parental consent is a requirement (Woods, Hagger &
McCormack, 2014). Shilling’s and Young’s review (2009) of parents’ experiences of being asked to
enter their child into an randomised controlled trial found that parents felt that experimental
treatment was superior (i.e., therapeutic misconception), suggesting that parents may construct trial
treatment as the answer to their child’s needs. Parents in my research may construct early
surveillance in this way because of the design of the program, in that children will be treated earlier
than if they were not part of the research, though clinical benefit is currently unknown. The
generally high recruitment rates in neonatology (Campbell, Surry & Royle, 1998), and paediatric
oncology (Ablett & Pinkerton, 2003) research, and indeed in early surveillance (99%) could be
indicative of parents’ constructions of research participation as directly benefitting their child
(Chudleigh, et al., 2013; Dixon-Woods, et al., 2008).
Lastly, evidence that attributing meaning of clinical benefit to early surveillance is a co-
constructed process was highlighted when reviewing understanding of early intervention among
parents. Whilst parents across both CF clinics acknowledged and made reference to keeping children
healthy through performing adequate levels of standard care, they also strongly identified with the
role of early intervention in their child’s long-term health outcomes. The following excerpts illustrate
parents’ value towards the opportunity for early intervention as a result of early surveillance:
Western Australian mother of a 12 month old baby - for me, it’s really important because
we want to know, we want to pick things up early and get onto it. That’s the whole
reason we came to WA, because we know that AREST CF was here, we knew that it was
a world-class program and we just want what is best for her, and I think the team can
really provide that.
Victorian mother of a 4 year old child - I mean it’s a weird thing because you feel like it‘s
a tough road to be on, but then there’s also the security of knowing that your child is
155 Parenting Children with Cystic Fibrosis
getting treated as soon as something comes up, so what you go through really is the best
thing for your child.
In reviewing the key health improvement strategies for children with chronic conditions
promoted by the Australian government, it is evident that early intervention is one of the primary
foci (Commonwealth Department of Health and Aged Care, 2000; Commonwealth Department of
Health and Ageing, 2009). Therefore, the current finding appears to reflect the dominant health
promotion discourse within this context. Accordingly, this finding illustrates that dominant discourses
may influence parents’ constructions of meaning in early surveillance. Collectively, findings from my
research revealed that constructs such as early intervention influence parents’ thinking about their
parenting behaviours and affects how they relate to their own experiences and understandings of
parenting within context of their child’s condition. Assumptions underlying an early intervention
approach highlight how policy and programs referring to such concepts can influence parents’ own
constructions of meaning in health promotion and thinking about what decisions they ought to
make. For this reason, it is important to be mindful of how certain discourses become the dominant
ones and to question whose needs they serve so that informed and unbiased decisions can be made.
To conclude this sub-theme, the therapeutic misconception is detrimental to an individual’s
understanding of research, which is crucial for an autonomous decision because it can undermine the
ability of the decision-maker to make an accurately-informed decision. If, indeed, the therapeutic
misconception is present for parents in my research, it means they may make inaccurate judgements
based on false beliefs as part of their process in appraising the research study. Based on the current
finding there is an urgent need to identify if strategies to avoid the therapeutic misconception are
required. Discussion now shifts focus to describe how cognitive dissonance can be experienced by
parents once the decision to participate in early surveillance has been made. Discussion about
cognitive dissonance will round out this theme by describing how early surveillance is interpreted by
parents as being in the best interest of their child’s health, but with some adverse psychological
consequences for parents.
5.7.2 Cognitive dissonance results in anxiety and/or ambivalence.
Western society promotes autonomy for medical treatment decisions and research
participation, however the consequences of such decisions are not as clear-cut as their idealities.
Psychology research has shown that agency and internal locus of control associated with
autonomous decision-making lead to a perceived sense of personal causality (Hashimoto & Fukuhara,
2004; Cobb-Clark, Kassenboehmer & Schurer, 2014; Pettersen, 1987). Research more specifically in
paediatrics (Botti, Orfali & Iyengar, 2009, Polizzi, et al., 2015), and in keeping with psychological
research on decision-making, shows that outcomes of decisions made by parents are internally
156 Parenting Children with Cystic Fibrosis
attributed to their control, thereby magnifying emotional response to an event as a result of a
parental decision. This means that accountability exists in an ethical decision that emerges from
parents’ demand of responsibility.
Some psychological consequences of parental decision-making about children’s medical
research participation reported by Botti and colleagues (2009) included increased grief, distress,
anxiety, guilt and blame, and ambivalence about autonomy, which resulted in decreased coping
ability. Consequently, psychological confusion, or cognitive dissonance, may manifest itself in
ambivalence and anxiety in decisional autonomy. For parents in my research, cognitive dissonance
was experienced acutely around the time of annual review, reaching its climax at the time of
relinquishing their child for medical procedures. Cognitive dissonance generally resulted in feelings of
anxiety or ambivalence about the decision to participate in early surveillance (Lavine, Thomsen,
Zanna & Borgida, 1998; Maio, Esses & Bell, 2000; van Harreveld, Rutjens, Rotteveel, Nordgren & van
der Pligt, 2009).
Cognitive dissonance is an emotional state occurring when two simultaneously held
attitudes or cognitions are inconsistent, or when there is conflict between belief and behaviour
(Festinger, 1962). Festinger’s (1962) cognitive dissonance theory focuses on how individuals strive
for internal consistency. When inconsistency (dissonance) is experienced, individuals largely become
psychologically distressed. Festinger conceptualised the existence of dissonance as psychologically
uncomfortable, which will motivate an individual to try to reduce the dissonance and achieve
consonance.
The presence of psychological discomfort for parents in my research as a result of cognitive
dissonance may transpire through two means. Firstly, cognitive dissonance may transpire because of
contradictory speculations about putting a child through a traumatic experience but for the greater
good of their health. For example, a Western Australian father of a 4 year old child stated, ’we think
its (early surveillance) important but there is that part of you that says "do we really need to be doing
this?” So it's a lot of questions that come up. But at the end of the day, we want to do what's right for
her. I suppose you just have a lot of questions that get raised in your own head.’ Secondly, cognitive
dissonance may transpire because the act (behaviour) of relinquishing a child to undergo a
(perceived) potential harm is contradictory to parenting responsibility of protecting children from
harm. For example, a Western Australian mother of a 6 year old child said, ‘I feel like my
responsibility as a parent is to protect my child from harm, but then I’m putting him in a threatening
and potentially harmful situation.’
For these reasons, cognitive dissonance could become a lived experience for parents in my
research. Echoed by Glasscoe and Smith (2011), they reported two frames of reference in making
decisions about a child’s CF treatment; self as mother, which referred to the intimate relationship
157 Parenting Children with Cystic Fibrosis
with her child and which was governed by emotional binding, and self as caregiver, which referred to
her professional role that was guided by logic and reason. Therefore, cognitive dissonance can create
negative psychological responses for the individual experiencing the dissonance (Elliot & Devine,
1994; Festinger, 1962; Harmon-Jones, 2000). For parents in my research, contradictory beliefs versus
behaviour generated anxiety whereas contradictory beliefs about early surveillance generated
ambivalence.
An example of anxiety caused by contradictory belief and behaviour came from a Victorian
mother of a 6 year old child who stated, ‘it just makes me feel anxious that I’m supposed to protect
her, yet something bad could happen because I put her in a dangerous and possibly harmful
situation.’ Contradictory beliefs can result in ambivalence towards a subject matter (Ben-Nun Bloom
& Arikan, 2011; Castro, Garrido, Reis & Menezes, 2009; Moody, Galletta & Lowry, 2014). A Western
Australian mother of a 3 year old child expressed ambivalence about her decision when she stated,
‘yeah, I was just unsure about it. A bit apprehensive really. I didn’t really want to do it but of course
you want to do it at the same time to make sure he’s ok. So yeah, I was just unsure about my decision
to do it and then to keep doing it.’
Ambivalence in psychological terminology refers to co-existence within an individual of both
positive and negative feelings towards the same person, object or action, simultaneously drawing
the individual in opposite directions (Lewin, 1939; 1943). Parents have positive feelings of power and
control over their child’s disease that eventuate from early surveillance results. They also have
negative feelings of lack of control and helplessness due to relinquishment of their child to threat
and potential harm, and possibly blame and guilt for their child’s traumatic, potentially dangerous
situation (Salgado, et al., 2011). The affective conflict about early surveillance leads to ambivalence
about the decision to participate.
Resolution of cognitive dissonance (i.e., consonance) is explained by cognitive dissonance
theory, which conceptualises attitude change from motivation to adjust attitudes to relieve cognitive
dissonance (Festinger, 1962; 1964). For the contradictory behaviour against the belief about threat of
harm, parents adjust their beliefs so as to align with their behaviour. In this sense, the behaviour of
relinquishing their child for surveillance of their chronic, often fatal illness is perceived as better than
the behaviour of not being involved in early surveillance. Another conception of this is that parents
would always choose to do whatever they could for the perceived benefit of their child’s health if it
was deemed to be safer than not doing anything. A Victorian father of a 4 year old child showed how
he adjusted his attitude to match his behaviour when he expressed that, ‘our goal is keep our kid
healthy. If that means putting her through some traumatic stuff with the purpose of keeping her
healthy and alive for longer, then that’s what we’ll do… so I just have to adjust the way I think about
it, which then makes me feel better about putting her through that kind of stuff.’
158 Parenting Children with Cystic Fibrosis
For the contradictory beliefs about trauma of early surveillance versus the greater good for
child and future CF generations, parents are in a continual cycle between the former and latter
beliefs. However, the belief in early surveillance as the best option for a child brings a sense of
cognitive resolution. For example, a Victorian mother of a 3 year old child expressed, ‘there's a lot of
guilt, but you know, deep down that when we decided to do the research, we know that it was for
good. For her benefit and obviously for future CF kids, so we said that it's going to benefit her so
that's what we try to keep always in our minds. That helps me to deal with our decision.’ If cognitive
dissonance is resolved, parents can still return to the cognitive dissonant state (Wicklund & Brehm,
1976), either as part of their continual evaluation of participation at each annual review or general
feelings of uncertainty.
Trust can function as a resolution to cognitive dissonance (Festinger, 1964). Partial or
complete elimination of cognitive dissonance may be a function of lowering expectancy that an
unfavourable or undesirable outcome will occur (Aronson, 1969). Trust functions to reduce possible
outcomes for a particular event because without trust, all contingent possibilities should be always
considered, leading to a paralysis of inaction (Braynov & Sandholm, 2002). A Victorian mother of a 4
year old child demonstrated how this occurs, ‘I'm thinking 'is she going to die?' Then I pull my
strength to thinking about 'that's irrational, what's the rational thought?’…I still feel, my heart is still
beating fast but it's all fine, I trust in the doctor’s abilities to look after my child.’ Therefore, it
appeared that trust allowed parents to relinquish their child to the medical team by enabling parents
to have confidence that their child was being taken care of. Moreover, it appeared that trust helped
parents to cope with relinquishment, expressed by a Western Australian mother of 4 year old child
who said:
well I think having trust in the staff helps me to cope with the procedure because I think
if you sit there, you start to think about things like 'she's having the tube put down her
throat, it could scratch the side of the oesophagus and then she might bleed,' so if you
tend to trust that what they're doing is the right thing, then you’re not worrying yourself
so much.
Application of the current finding to Hall’s (2002) conceptual model of trust within a
healthcare setting highlights importance of perceived competence of physicians for parents in their
capacity to trust medical staff. The notion of competence is central to trust within a medical setting,
and is divided into interpersonal and technical competence (Hall, et al., 2002). Competence is the
possession of required adequacy in a given situation (Reber & Reber, 2001), and so applied to
interpersonal competence within a medical setting, is the perception parents have about the
adequate nature of a physician's interpersonal skills and demeanour (Hall, et al., 2002). A Victorian
mother of a 4 year old child expressed this element of trust when she said, ‘trust has a lot to do with
159 Parenting Children with Cystic Fibrosis
it…I mean, you get to know them personally, one on one. So when they come up to you and say such
and such is going to happen and you look at them in the eye and you go 'yep right, ok.' Technical
competence is viewed as having good practices, making correct decisions, avoiding making mistakes
and producing the best achievable outcomes possible (Hall et al.), and is generally evaluated through
considerations of physician experience, thoroughness and knowledge. This assessment is mainly
based on patient experiences with the clinician. A Western Australian mother of a 5 year old child
described this element of trust when she stated, ‘I kind-of think that they do this every day so you
just have to trust their judgement.’
To conclude this sub-theme, simultaneous opposing attitudes, values, beliefs and behaviours
can result in cognitive dissonance, which can result in anxiety and/or ambivalence about parents’
decision to involve their child in early surveillance. Cognitive dissonance, and its resulting anxiety
and/or ambivalence, appears a parental compromise for the perceived beneficial, long-term child
health outcomes of early surveillance participation. Trusting in medical and research staff can help
parents to manage psychological adversity associated with their child’s early surveillance
participation. Therefore, developing and maintaining a therapeutic relationship comprising physician
competence and parental trust is of paramount importance given the reciprocal and continual
nature of the therapeutic relationship for ongoing care for children with CF.
In review, the decision to participate in early surveillance for CF lung disease is primarily made
on the premise that it will directly benefit a child’s clinical health outcomes, resulting in transcending
feelings of hope for their child’s future. However, a salient parental outcome from the decision to
participate in early surveillance is cognitive dissonance, which can result in anxiety and/or
ambivalence about the decision. Parents are willing to deal with the such psychological adversities
associated with early surveillance as a compromise for the perceived direct benefit to their child and
the larger CF community; this is how early surveillance is believed to be in the best interest of the
child, but my research has shown, notwithstanding psychological adversity for parents.
The final, transcending interpretation of the lived experience for parents whose children
undergo early surveillance for covert CF lung disease is an assertion of hope. Hope for the future.
Hope through participation in early surveillance transcends all themes, and is therefore interpreted
as an over-arching and ever-lasting structure of parents’ lived experience. Whilst parents did not
speak explicitly about hope, its placement at the end of the findings chapter allows me to convey its
transcendence in terms of its essence to the lived experience of parenting children with CF in the
context of early surveillance. It allows me to leave the reader with the sense of transcendence with
which it exists for parents. That is, hope for a child’s future, for future CF generations, and for a cure
are the primary outcomes of early surveillance for parents. The sense of hope transcends parents’
experiences, emotions, cognitions, and outcomes associated specifically with early surveillance,
160 Parenting Children with Cystic Fibrosis
therefore substantiating that early surveillance is perceived by parents as being in the best interest of
the child.
5.8 Construction and conceptualisation of hope
When an infant is diagnosed with CF, without hope for their future there seems little point
of vigilance in treatment and health awareness, and little point of sometimes struggling and
sometimes embracing life with CF. However, from devastation of a CF diagnosis rises hope for a
child’s life; hope is primarily for a child’s future in terms of longevity, quality, normality and health.
According to the Ortony, Clore and Collins Classification System of Emotions (1990), hope is
a primary emotion that refers to feeling pleased about the prospect of a desirable event; it is a
positive attitude based on an expectation of positive outcomes related to events and circumstances
in one's life or the world at large. Hope has been linked to the perceived availability of successful
pathways related to goals, combined with a determined plan for reaching goals along with a
perception of successful agency related to the goals (Synder, et al., 1995).
AREST CF provides a goal-oriented positive orientation towards the future (Cutcliffe & Herth,
2002; Samson, et al., 2009); it offers parents a pathway to achieve their new end goal after a child’s
diagnosis irrevocably changes initial parent and family goals. AREST CF offers a window of
opportunity, of hope, to treat a child as early as possible. The notion of earliest surveillance leading
to earliest possible treatment available in the world is perceived by parents as the best chance their
child has at avoiding morbidity and achieving increased life expectancy, which creates hope for a
child’s future. Therefore, early surveillance for CF lung disease is seen by parents as a successful
pathway for their new goals that includes a perception of successful agency related to such goals.
That is, the sense of control created by early surveillance acts in both existential and practical terms
in reaching parents’ alternative goals for their child created as part of the new normal after
assimilation of diagnosis into the family. Moreover, agency has been reported as a significant factor
in predicting subjective well-being in parents of children with special needs (Shenaar-Golan, 2015).
As AREST CF provides a pathway to achieving new goals and a sense of agency towards achieving
them, it may well contribute to their subjective well-being.
Parents’ construction of hope for a child’s future – their lifeworld, occurs through their
beliefs in treatment as the best option because of perceived direct clinical benefit to the child health
outcomes generating assurance in child health. Recall that hope for a child’s future could be
constructed early on when uncertainty in morbidity and mortality are understood (in sub-theme
Understanding CF). Hope can then become crystallised over the first few years in early surveillance
through belief that early surveillance and subsequent treatment will provide direct clinical benefit.
Uncertainty is a precondition for hope, as hope is about possibilities (Verhaeghe, van
Zuuren, Defloor, Duijnstee & Grypdonck, 2007). As early surveillance functions to reduce
161 Parenting Children with Cystic Fibrosis
perceptions of uncertainty about disease progression, it may moderate the relationship between
hope and uncertainty. That is, early surveillance may decrease uncertainty, which could increase
hope. Additionally, hope in the face of uncertainty contributes to adaptation (Samson, et al., 2009;
Truitt, Biesecker, Capone, Bailey & Erby, 2012), suggesting that positive parental outcomes from
early surveillance (i.e., increased hope, reduced uncertainty, agency and perceived control) may
function as moderators of adaptation and effective coping, which could ultimately influence
resilience.
Whilst it is a complex concept, there is a consensus that hope has a role in helping
individuals to cope with existential challenges, including the ability of a parent to cope with their
child’s life limiting disease (Samson, et al., 2009). Hope has been reported as a protective factor
against psychological distress in parents of chronically ill children (Barlow & Ellard, 2006; Ogston, et
al., 2011), functioning as a moderator between stress and maladjustment, suggesting a buffering
effect against maladjustment (Horton & Wallander, 2001). Based on outcomes such as these, hope is
understood to provide the basis for resilience (Petersen & Wilkinson, 2015).
Whilst parents in my research fluctuate between positive and negative outlooks on life,
regardless of parents’ transitory state of being, their hope never fades. All adverse parental
experiences and outcomes that result from early surveillance are transcended by the hope they
sense from early surveillance for their child’s long-term health. That is, adverse parental experiences
associated with early surveillance are a by-product of hope for long-term child health, which can
reduce parental anxiety and emotional impact of CF (Wong & Heriot, 2008). In context of my
research, it appears that hope afforded by participating in early surveillance functions to reduce
psychological distress associated with parenting a child with CF. The interpretation of hope as a lived
experience for parents whose children undergo early surveillance supports the notion of short-term
parental adversities caused by early surveillance (e.g., fear and helplessness at relinquishment, guilt
from negative annual review results, anxiety and ambivalence from cognitive dissonance) as a
parental compromise for long-term child, parent, family, and CF community benefit.
Knowledge is the outcome of early surveillance for CF lung disease; about a child’s current
health status and also about CF disease progression in early childhood. Being part of a program that
is the first in the world to examine the earliest signs of lung disease in young children with CF,
parents not only carry hope for their child but they also carry hope for future CF generations. In
terms of hope for future CF generations, parents are contributing to science through research, which
they perceive as hope for children in the future through their contribution to research now.
Ultimately, there is hope for a cure for CF, however this more global element of parents’ hope is
secondary to the direct hope for their child’s future.
162 Parenting Children with Cystic Fibrosis
Knowledge is power. Power affords hope. Therefore, hope is the essential meaning of early
surveillance for parents. Hope for the future, child, CF community - a cure. The diagram below
depicts processes and factors for parents’ construction of hope. The diagram illustrates how belief in
early surveillance as the best treatment option because of perceived direct clinical benefit generates
perceptions of improved child health, which creates a sense of assurance in child health. Assurance
in child health from improved health outcomes facilitates hope for their child’s future.
Figure 10. How hope is constructed by parents
Hope for the future is a both a product of, and transcends, CF and early surveillance. In a
medical context, hope is “strongly linked to the consumption of medical technologies, particularly
therapies that promise to offer cures,” (pp. 115) (Petersen & Wilkinson, 2015). Whilst early
surveillance for CF lung disease does not claim to offer a cure, dramatic technological advances in
medicine, such as those offered by AREST CF, offer more potential opportunities for hope for
recovery than were available in the past (Gengler, 2015). In context of my research, hope is for
improved health outcomes and hope is for future CF generations. Ultimately, hope is for a cure. As a
dominant parental outcome of early surveillance, hope is interpreted as a lived experience for
parents of children undergoing early surveillance of CF lung disease. Hope appears to overcome fear
and reduce stress, which likely contributes to resilience; and it appears to develop from perceived
163 Parenting Children with Cystic Fibrosis
control over disease progression afforded to parents by AREST CF providing a sense of existential
and practical agency to achieve new end goals.
5.9 Summary and conclusion
The underlying aims of this chapter were to describe how parents in Western Australia and
Victoria whose children have CF constructed meaning and understanding about the experience of
parenting within the context of early surveillance for CF lung disease in the first 7 years of life, and
how they cope. Five major themes emerged and were explored, as were their respective sub-
themes, these were, 1) Redefined Expectations and Reimagined Identity; Redefined Reality and the
sub-themes of Collapsed and Redefined Expectations, and New Normal, 2) Understanding the
Unknown and Understanding Uncertainty and the sub-themes of Understanding CF, and
Understanding Early Surveillance for CF Lung Disease, 3) Good Days and Bad Days; Fluctuation
Between Positive and Negative Outlook on Life and the sub-themes of Bad days, Good Days, and
Fluctuation Between Good and Bad days, 4) Annual Surveillance is a Significant Event and the sub-
themes of Indicator of Treatment Efforts, Parental Outcomes Directly From Early Surveillance, and
Fear From Diagnosis Exacerbated Each Annual Review can Result in Anxiety, and 5) Early Surveillance
is in the Best Interest of My Child with sub-themes of Perceived Clinical Benefit to Child Health and
Cognitive Dissonance Results in Anxiety or Ambivalence. Evidence was provided for each theme
through the provision of examples drawn from the data and linked with the literature. Lastly, hope
for the future was interpreted as an over-arching and pervading parental outcome from their child’s
early surveillance.
There is much empirical and anecdotal evidence regarding adjustment and adaptation, or
normalisation, for parents of newly diagnosed infants (Glasscoe & Smith, 2011; Hodgkinson & Lester,
2002; Hopkins & Gallo, 2012; Knafl, et al., 2010; Ray, 2002). Diagnosis of CF via NBS and its
treatment become assimilated into family life, and parents’ experiences from my research provide
further support for the importance of adaptation for long-term parent mental health and family
functioning, and child physical health. However, the most salient finding from parents’ experiences
of their child’s CF diagnosis related to the collapse of child, parent and family expectations and
rebuilding expectations and parental identity, which were commonly discussed outcomes for
parents in my research. These experiences also affected the way parents viewed their child’s and
family’s future by influencing their expectations of family life both in both positive and negative
ways. This was particularly evidenced by parents recalling negotiation from devastation of diagnosis
to acceptance of their new way of life.
In addition, both the parental role and parental identity were reconstructed to include the
carer who administered intensive and time-consuming treatment. In particular, parents appeared
vigilant about administering treatment, which resulted in high self-efficacy for treatment
164 Parenting Children with Cystic Fibrosis
management. Although some of the above factors led to early adverse responses and reactions,
parents described how support from medical staff and from family and friends, along with time to
develop understanding and experience of treatment management, contributed greatly to their
negotiation of parenting a child with CF. Support was identified as important to parents’ experiences
of diagnosis and treatment management, and highlights the importance of drawing attention to
available support mechanisms and resources. It also raises awareness of those parents without
family support or who are geographically isolated may struggle to cope effectively and might
encounter barriers to successful management, adjustment and adaptation.
Despite the emphasis of treatment management of paediatric CF primarily focussing on child
health outcomes, findings from my research support that a more holistic approach to caring for
children with CF is needed that takes into account the well-being of the family. For example, parents
frequently referred to intra-psychic processes that resulted from becoming primary carer for their
child. Undervaluing these psychological aspects important for treatment management restricts
possibilities for addressing the more holistic needs in the promotion and maintenance of health and
well-being, and potentially limits opportunities for the families’ adaptation and resilience. Moreover,
findings from my research offer insight based on parents’ lived experiences that can be used to
improve delivery and implementation of future policy and practice for diagnosis of CF via NBS and its
management in the early years of life.
Another salient finding from my research indicated that constructed thoughts about early
surveillance for covert CF lung disease at both sites were primed by biomedical discourse used to
describe their early surveillance motivations. Findings particularly highlighted that constructs such as
disease progression and early treatment were well-recognised by parents, as well as influencing their
health beliefs and behaviours towards their children. However, though parents’ initial comments
about their experiences of their child’s early surveillance frequently related to adverse experiences
and outcomes, their experiences also reflected that the positive psychological factors influencing
their experience of early surveillance were of equal, if not greater significance. This was evidenced
by the volume of parents’ discussions focussing on beneficial long-term outcomes associated with
early surveillance. Significantly, these factors affected perceptions of themselves as parents and the
future of their child.
A particularly noteworthy finding from my research was that it was more common than not
that parents expressed feelings of hope and control when reflecting on their early surveillance
experience. Feeling hopeful and in control because of, and grateful for, their experience was a
common way for parents to express how they attributed meaning to early surveillance and
ultimately influencing their appraisal of their child’s condition and their early surveillance experience
itself. Despite the challenges faced by parents, findings from my research indicate that parents had
165 Parenting Children with Cystic Fibrosis
good emotion regulation. This was primarily evidenced by their maintenance of a positive attitude
expressed through acceptance of change resulting from their child’s diagnosis and the regulation of
the dynamics between adverse and beneficial outcomes of early surveillance.
Moreover, how parents constructed meaning of their early surveillance experience
appeared to be primarily mediated through temporal factors mostly reflected in context of gaining
knowledge and experience over time, which facilitated understanding. Making comparisons with
others (i.e., children, families) also allowed parents to construct meaning about their early
surveillance experience, as well as provided a way of thinking about, and planning for their child’s,
and family’s futures. In addition, the notion of gratitude was found to mediate participants’
constructions of meaning around their early surveillance experience with parents using it to create a
perception of appreciation, enabling them to maintain a sense of indebtedness for their early
surveillance experience. Lastly, support from medical and research staff, family and friends was a
primary coping resource, permitting successful management of practical and psychological aspects
of parenting a child with CF in context of early surveillance.
Therefore, findings from my research support the notion that the early surveillance
experience is not an isolated event; that it permeates the lives of parents and their families who
experience it. These findings suggest that constructions of early surveillance and meanings
attributed to it influence constructions of CF and its management. However, insight from my
research also highlights the psychological and social content of parents’ experiences that were of
equal, if not greater significance to their experience of early surveillance. Importantly, findings from
my research provide a particularly strong foundation for the centrality of hope, existential belief
systems and personal meaning orientations to the early surveillance experience. Isolating parenting
and family processes described in this chapter, and identifying how they might function together to
explain, and predict, family outcomes of managing CF is fulfilled in the next chapter by applying the
research findings to the The Resiliency Model of Family Stress, Adjustment and Adaptation
(McCubbin & McCubbin, 1993).
166 Parenting Children with Cystic Fibrosis
CHAPTER 6
Parenting a child with CF in the context of early disease surveillance: Adapting to paediatric chronic disease from a theoretical perspective
This chapter presents further discussion of the research findings, with particular reference to
applicable theoretical frameworks through which to view the findings. Psychological factors
identified in the preceding chapter that may contribute to adaptive coping within families are
applied to the Resiliency Model of Family Stress, Adjustment and Adaptation. How the model is
applicable to families whose children with CF undergo early surveillance is speculated, along with
how some psychological factors that may contribute to coping can be negatively influenced by the
early surveillance experience. By applying the research findings to the Resiliency model,
considerations are made for how constructions of experience and attributions of meaning may
contribute to coping with lived experiences. Such considerations demonstrate how parents might
adapt to parenting children with a chronic condition who undergo early surveillance for covert
disease progression.
167 Parenting Children with Cystic Fibrosis
Resilience is accepting your new reality, even if it’s less good than the one you had before.
You can fight it, you can do nothing but scream about what you’ve lost, or you can
accept it and try to put together something that’s good…Elizabeth Edwards
6.1 Introduction
As theoretical models are developed from previously tested knowledge and identify a plan
for interpretation of findings (Bogdan & Bilken, 2006), they provide a background that supports
explanation of phenomena that includes concepts and relationships intended to be interpreted. In
this way, theoretical frameworks guide research findings, provide a foundation for their application,
and can drive further exploration and analysis. Therefore, theoretical frameworks advance
understanding of research findings by being able to apply findings beyond their current context as
well as providing an opportunity to support or refute a model’s components within the findings’
current context.
The Resiliency Model of Family Stress, Adjustment and Adaptation (McCubbin & McCubbin,
1993) provides a structural guide for exploring and identifying relevant variables of interest and their
potential associated relationships and outcomes as analysed and interpreted in the previous
chapter. As such, concepts within the Resiliency Model may begin to indicate how parental
processes can influence, and be influenced by, systemic, family and personal factors that in turn
influence parental coping of, and ultimately adaptation to, CF specifically, and paediatric chronic
illness more generally.
The current chapter widens the scope of the research findings and their interpretation as
they might apply within the family context. In this way, the chapter provides a stepping stone to
broader applicability of the research findings, not only to the family context but to the theoretical
advancement of the Resiliency Model. Lastly, the chapter argues for the use of the Resiliency Model
with families whose children undergo early surveillance for CF lung disease for therapeutic
intervention as well as further research.
6.2 Application of Resiliency Model of Family Stress, Adjustment and Adaptation
The Resiliency Model of Family Stress, Adjustment and Adaptation (McCubbin & McCubbin,
1993) attempts to explain why some families recover and are deemed resilient whilst others remain
vulnerable and some deteriorate under the same circumstances. Family resiliency can be defined as
“positive behavioural patterns and functional competence that individuals and the family unit
demonstrate under stressful or adverse circumstances, which determine the family’s capacity to
recover by maintaining its integrity as a unit whilst ensuring, and where necessary restoring, the
well-being of family members and the family unit as a whole (McCubbin, Thompson, & McCubbin,
1996, pp. 5)”. Given the observation that families in my research generally appeared to cope
168 Parenting Children with Cystic Fibrosis
successfully with a psychologically and logistically challenging situation, a strength-based approach
to understanding how parents manage their child’s CF is appropriate.
As adaptation to a paediatric chronic condition is the most desired outcome for families, the
adjustment phase of the model will not be a focus of the current chapter (refer to Figure 1 in
Chapter 3 for a diagram of the adjustment phase). As such, the adaptation phase of the Resiliency
Model serves as a framework to view how parenting processes and outcomes may contribute to
family resiliency in the context of my research. Moreover, the model lends itself to recognising how
early surveillance might contribute to those processes identified in the model as important factors
for family adaptation and resiliency. By applying my research findings to the Resiliency Model, this
chapter presents how processes and outcomes of parenting a child with CF in the context of early
surveillance might contribute to adaptation to paediatric chronic disease. See the following page for
a diagram of the adaptation phase of The Resiliency Model of Family Stress, Adjustment, which the
remainder of the chapter addresses.
The Adaptation phase of the model begins by recognising that an abundance of stressors,
strains and transitions impact upon the initial family crisis situation. However, due to the nature of
my research, such factors were not identified. It is therefore acknowledged here that parents and
their families are likely to have a multitude of historical, economical, developmental and personal
difficulties that can influence the way a family initially experiences a crisis situation. Once the family
crisis situation has occurred (i.e, for families in my research, it is the diagnosis of CF), parents start to
institute new patterns of family functioning. My thesis will apply the research findings to the
Resiliency Model from this point on, highlighting factors and relationships the model deems relevant
for adaptation to paediatric chronic disease.
169 Parenting Children with Cystic Fibrosis
Figure 11. Adaptation phase of The Resiliency Model of Family Stress, Adjustment and Adaptation. Adapted from “Families, Health and Illness: Perspective on Coping and Intervention,” by H. I. McCubbin and M. A. McCubbin, 1993, p. 23. Copyright 1993 by Mosby.
By applying the research findings to the Resiliency Model, considerations can be made for
how constructions of experience and attributions of meaning identified in the previous chapter may
contribute to coping with lived experiences of parenting children with CF who undergo early
surveillance for lung disease. Applying the research findings to the Resiliency Model enables
identification of essential interacting components of family life and can explain, as well as predict,
family processes and outcomes of managing a paediatric chronic condition. The following page
outlines how constructs and processes identified in the previous chapter might function together to
explain, and predict, parent and family outcomes of managing a paediatric chronic condition,
specifically in the context of early surveillance for CF lung disease. The remainder of the chapter will
detail the diagram’s constructs and relationships for adapting to CF in the context of early
surveillance from the point of establishing family patterns following diagnosis (i.e., the crisis
situation).
170 Parenting Children with Cystic Fibrosis
Figure 12. Research findings applied to The Resiliency Model of Family Stress, Adjustment and Adaptation
171 Parenting Children with Cystic Fibrosis
6.2.1 Newly Instituted Patterns of Functioning.
Families have a wide range of established patterns of functioning that offer stability to
family life which, according to the model (McCubbin & McCubbin, 1993), may not be adequate to
manage illness within the family system. Therefore, newly instituted patterns are needed to keep
the family functioning with stability while managing the illness and its related hardships. The major
changes in the family patterns of functioning found in my research were; establishing a home
regimen for the child’s treatment management, and redefining present and future expectations
about parenting and family life. Essentially, the process of normalising the condition and its
treatment represents a newly established pattern of family functioning that re-establishes family
stability and resources, thereby promoting adaptation in families.
Re-establishing family patterns after introduction of CF is not achieved overnight. Trial and
error practices and strategies must be negotiated and compromised to fit within the family’s
already-developed schema (i.e., blueprint) for their family functioning that has evolved over time
(McCubbin & McCubbin, 1993). By establishing both practical and existential patterns of functioning
through normalisation and redefinition, parents are able to reaffirm their family that constitutes a
redefined sense of reality including managing a child’s chronic condition. How effective these newly
established patterns of family functioning become is determined by the degree to which they are
congruent with the family’s schema (McCubbin & McCubbin, 1993), discussed shortly.
By assessing changing family patterns, HCPs can determine strategies for intervention by
distinguishing adaptive and destructive patterns of functioning that are tailored to address illness-
specific and related hardships. Assessing both new and modified patterns of functioning can identify
what these families deem important to them, and the strengths and resources they have. Moreover,
assessing family functioning when faced with their child’s chronic condition can begin to shed light
on what meaning families attach to themselves, the illness and their future (McCubbin & McCubbin,
1993).
Normalising CF management in practical terms and normalising both CF and its management
in existential terms, influences all other components identified in the Resiliency Model as important
factors for adaptation. Therefore, these patterns of family functioning contribute to what resources
a family has and chooses to draw upon. For example, maintaining a home treatment regimen
enables parents to develop a high sense of self-efficacy for keeping their children healthy. Self-
efficacy is a family resource that is influenced by newly instituted patterns of family functioning. At
the same time, self-efficacy influences a family’s coping patterns. Similarly, established patterns of
family functioning contribute to the level of social support families draw upon and accept as part of
their adaptation. For example, successful normalisation and acceptance of CF into family life is likely
172 Parenting Children with Cystic Fibrosis
to enable parents to draw upon social support that further facilitates, as well as validates, their
adaptation strategies and outcomes.
A family’s situational and family schema appraisals are also influenced by what patterns of
family functioning parents establish as part of their aim to achieve adaptation to their child’s chronic
condition. Moreover, early surveillance becomes part of the newly established patterns of
functioning. As such, early surveillance becomes part of the fabric for how a family will adapt to their
child’s CF. Accordingly, early surveillance contributes to development, establishment and
maintenance of the remaining components identified within the Resiliency Model as important
factors for adaptation.
6.2.2 Situational and family appraisal.
The Resiliency Model (McCubbin & McCubbin, 1993) emphasises two levels of family
appraisal that play a role in a family’s ability to manage a crisis situation, which shapes the course of
family adaptation. Firstly, situational appraisal of the family’s capability is the critical relationship
between the illness-related demands on the family and their capabilities and strengths to manage
these demands. The family’s situational appraisal would reveal their in/adequacy to manage the
situation and serve as the basis for additional coping or changes in family patterns of functioning.
Why the family’s situational appraisal becomes crucial in shaping the family’s response to illness is
that through the process of situational appraisal (including evaluation of the illness, assessment of
family capabilities and strengths, and evaluation of alternative courses of action and coping
strategies), the family ultimately comes together as a unit to cope with the illness.
6.2.2.1 Situational appraisal.
By applying my research findings to the Resiliency Model, parents appraise their family’s
situation by evaluating CF in the context of early surveillance. The inherent link between the two
brings a range of emotions for parents including fear and chronic sorrow. However, parents were
also able to experience altruism and gratitude and perceived control and power that came from
early surveillance participation. Other parents of children with CF have also reported both positive
and negative appraisals of their child’s condition (Baldaccino, et al., 2012), however their appraisals
were not in the context of early surveillance. It appears that for parents in my research, early
surveillance outcomes seemed to off-set both the early surveillance experience and negative
responses to having a child with CF.
Cystic fibrosis and early surveillance certainly place significant burden on parents, their
children and their families. They both demand psychological and practical attention from parents
that can impact upon families. However, CF within the context of early surveillance provides parents
with strength that seemed to transcend CF and early surveillance. For example, early surveillance
173 Parenting Children with Cystic Fibrosis
was appraised as a family resource in terms of the knowledge acquired on an annual basis about a
child’s health. Parents were able to use both their child’s early surveillance results and the early
surveillance principles as sources of family strength. That is, knowing of their child’s health and
knowing they are contributing to something greater were sources of strength for parents. Therefore,
whilst parents are placed under additional burden of early surveillance, the transcendent strength
provides a perception of adequacy in managing and coping, both with CF and early surveillance.
Social comparison appraisals at this level that function as sources of family strength for coping with
adversity are discussed in the section: family resources – strengths and coping strategies as family
capability.
6.2.2.2 Family appraisal.
According to the Resiliency Model, the other level of appraisal that contributes to a family’s
ability to manage a crisis situation is family appraisal, consisting of schema and meaning. When
faced with an illness-induced family crisis that demands changes in organisation and patterns of
family functioning, the family is required to appraise its past and its future to give meaning to the
illness and to the resulting changes within the family system. This meaning can facilitate adaptation
(McCubbin & McCubbin, 1993).
A family’s schema consists of value attached to shared or accepted goals, priorities and
expectations that have been developed and shaped over time. The family schema, unlike other
components in the Resiliency Model, is relatively stable and it is used as a major reference point
against which situational appraisals are contrasted and shaped. The family schema is more stable
and enduring than situational appraisals because it captures the family’s values and internal sense of
identity and goals.
As part of establishing and implementing new patterns of functioning, the existential (as
opposed to practical) normalisation of CF into the family created changes in family schema.
Appraisal at this level included assessing and redefining goals, priorities and expectations that were
congruent with introduction of CF into the family system to give meaning to the illness and to the
changes made. Family schema appraisal is posited as the most stable component in the model
(McCubbin & McCubbin, 1993). Changes at this level contributed to disintegration of previously-
established family goals, priorities and expectations, which initially created instability and
uncertainty about the family’s future goals and expectations.
The situational appraisal of a child’s condition within the context of its unique management
gave parents a new reference point to re-establish goals, priorities and expectations that matched
their newly-established patterns of family functioning. That is, early surveillance as a family source of
strength functioned as a framework to redefine goals, priorities and expectations associated with
174 Parenting Children with Cystic Fibrosis
their child’s CF. Being part of a unique treatment program, and gaining information about a child’s
health, enabled parents to give meaning to their required family changes because of their
involvement in the early surveillance program. Consequently, family schema was redefined within
the context of early surveillance to give meaning to CF and to the changes made. Whilst fostering
family coping and legitimising changes in family functioning, the family’s schema has as one of its
central functions the development of family meanings (McCubbin, Thompson, & McCubbin, 1996).
6.2.3 Meaning-making.
Due to required changes to family goals, priorities and expectations, as well as changes to
the family’s pattern of functioning due to a child’s chronic condition, the family’s schema appraisal
plays a central role in how families give meaning to the illness and the family’s changes. This level of
appraisal functions to develop congruency between changes in the family’s schema and their
instituted patterns of family functioning. Developing a sense of family meaning to changes created
by a family crisis is a difficult and demanding process, achieved only through perseverance, patience,
negotiation and shared commitment. Therefore, meaning-making takes on critical, and some would
argue central, importance in how families cope with their child’s chronic condition, ultimately
contributing to family adaptation.
Parents take on new roles as primary carers of the sick child and develop new patterns on
family functioning around CF management. At the time of diagnosis, parent struggle to make sense
of, and give meaning to, their child’s condition because the family’s original family schema is
threatened. Therefore, they battle to give meaning, and thus legitimacy, to the new parental roles
and family patterns. Over time, and with input from early surveillance, parents are able to introduce
new meaning to the changes they initially struggled with.
Parents were able to experience perceptions of power and control over their child’s
condition because of their participation in early surveillance through knowledge and ability to act on
findings. Perceived power and control facilitated a sense of gratitude for the opportunity to
participate in early surveillance. Perceived control and power over disease progression, along with a
sense of gratitude for early surveillance, combined to give parents the meaning of hope; hope for
their child, hope for their family, hope for the future. These new meanings, which do more than
explain the illness, appeared to bring legitimacy and value to the family changes and seemed to give
parents a sense of purpose and meaning beyond the illness itself. With this, the overall changes in
the family unit gain both meaning and acceptance, and seemed to be the basis of the family’s
emerging sense of calm and commitment.
As family meaning fosters congruency between newly instituted patterns of functioning and
family schema appraisal, it appeared that hope from early surveillance facilitated congruency
175 Parenting Children with Cystic Fibrosis
between normalisation of CF and its treatment (inclusive of early surveillance practices) and parents’
redefined goals, priorities and expectations for their family. Hope is a mediator between a family’s
actions and beliefs: families act in a certain way because of the belief it will benefit their child – and
that belief is facilitated by what they are told the early surveillance program will do for their child.
That is, hope mediates the relationship between normalisation of CF and its management in the
context of early surveillance and a family’s redefined future expectations.
Newly instituted patterns of family functioning are the most predictive component within
the Resiliency Model of un/successful coping because they are foundational to all other processes
that follow (McCubbin & McCubbin, 1993). Family meaning must match new patterns of family
functioning for adaptation to be likely. Parents in my research were able to create congruency
between changes in family patterns to accommodate CF in their daily lives, and the meaning
attributed to those changes. Participating in early surveillance for CF lung disease (i.e., changes in
family patterns) empowered parents through information acquisition and the ability to act on the
findings, which seemed to function as a mechanism for congruency between changes in family
behaviour arising from CF diagnosis and the meaning created from those changes. Parents
attributed hope to their child’s’ future from empowerment afforded by early surveillance. Whilst the
Resiliency Model posits the reciprocal nature of newly instituted patterns of functioning, family
schema and situational appraisal, and family meaning-making in adapting to chronic illness within
the family, my research findings have shown how these elements of the Resiliency Model might
function to facilitate coping with, and adaptation to, CF in the context of early surveillance.
6.2.4 Family resources – Strengths and coping strategies as family capability.
McCubbin and McCubbin (1993) define capability as the potential a family has for “meeting
all of the demands it faces” (pp. 45). They emphasise two types of capabilities; resources that a
family has, and coping behaviours and strategies, which is what a family does as individual members
and as a unit. Whilst the model identifies individual, family and community resources, my thesis will
focus on individual resources from the perspective of the parent. Adaptive resources are
characteristics that facilitate adaptation (McCubbin & McCubbin, 1993). A number of resources
highlighted in the family stress literature as critical for meeting demands during illness within a
family (Clark, Gong & Kaciroti, 2014; Fink, 1995; Knafl, et al., 2013; Tak & McCubbin, 2002;
Wallander, Varni, Babani, Banis & Wilcox, 1989) have been found in my research. Firstly, acquiring
knowledge and skills through education, training and experience means that family members can
perform tasks with greater efficiency and ease. Knowledge also helps parents to understand why
tasks associated with illness management are necessary. Acquiring knowledge itself about their
176 Parenting Children with Cystic Fibrosis
child’s condition is another component of this family resource because knowledge in this instance is
seen by parents as a form of power.
From a philosophical perspective, knowledge is an epistemological matter. In the current
context, knowledge creates a sense of control over CF – it appears that knowledge is power.
Foucault’s (1980) popular theorising of the complex relations between knowledge and power
provide a structural approach to understanding how the mechanisms of knowledge and power can
create a sense of control. He asserted that; power is not a thing but a relation with knowledge,
power increases capacity to control, and control from power is not simply repressive but it is
productive. Theory and research both attest to a positive association between knowledge thorough
education and a sense of control (Ross & Sastry, 1999; Pallas; 2000; Schieman & Plickert, 2008).
Notwithstanding the psychological adversity of gaining knowledge about a child’s disease
progression, parents are able to subjugate their child’s condition that has been portrayed to them as
an uncontrollable entity. Moreover, a sense of control shares conceptual ground with self-efficacy
and internal locus of control (Rotter, 1966), which are salient attributes for parents in my research.
Therefore, belief in the ability to conduct home treatment (i.e., self-efficacy); accountability for child
health (i.e., internal locus of control) and a sense of control over disease progression combine to
create this sense of power over their child’s condition, which, according to the Resiliency Model, acts
as a resource that positively contributes to a family’s coping patterns.
Secondly, a sense of mastery is an important adaptive resource for coping with paediatric
illness; it is the belief that the actions one performs will contribute to control over the circumstances
in one’s life (Reber & Reber, 2001). Based on definitions in the paediatric medical literature, my
thesis uses this term to refer to parents’ perceived competency and subsequent confidence in their
ability to achieve the required home treatment regimen. Achieving the home treatment regimen
gives parents a sense of mastery, which supports parental self-esteem. However, in context of my
research, sense of mastery can be supported or disrupted by early surveillance results (i.e.,
un/favorable review results), which in turn can affect parental self-esteem.
Lastly, self-esteem is an adaptive resource (McCubbin & McCubbin, 1993). It is a positive
judgement about one’s self-worth (Reber & Reber, 2001). For parents in my research, self-esteem
emanated from their competency and confidence in being able to conduct home treatment
adequately. Therefore, sense of mastery is influenced by how well parents can establish and
maintain CF management. Moreover, early surveillance results are constructed by parents as an
indication of how well they have been conducting home treatment, which therefore, means that
early surveillance results can either support or disrupt development and preservation of self-esteem.
177 Parenting Children with Cystic Fibrosis
Sense of mastery and self-esteem have been highlighted by many researchers of family
stress as critical factors in the family stress process because they are essential for effectively
managing demands (Folkman, Lazarus, Gruen & DeLongis, 1986; Hobfoll & Spielberger, 1992; Raina,
et al., 2005). Therefore, an important mechanism that may link stress to negative family outcomes is
the weakening of self-esteem due to failure of mastery (Cantwell, Muldoon & Gallagher, 2014;
Pearlin, Menaghan, Morton & Mullen, 1981; Silver, Bauman & Ireys, 1995). Perceived mastery of CF
management is affected by early surveillance because unfavourable review results can undermine
parents’ perceptions of their ability to conduct home treatment, resulting in adverse effects on their
parenting self-esteem.
Accordingly, the family coping resources (as well as constructed relationships between
them) of acquiring knowledge and skills, sense of mastery, and self-esteem can be influenced both
by a family’s capability for CF management and by early surveillance. Therefore, acquiring
knowledge from early surveillance about a child’s condition is an adaptive mechanism for coping
with CF. However, depending on what the early surveillance results yield for parents, sense of
mastery and self-esteem can either function as coping mechanisms for coping with CF or they can
hinder parents’ ability to cope effectively.
Coping behaviours and strategies are identified by the Resiliency Model as what a family
does as individual family members and as a system to meet the demands it faces (McCubbin &
McCubbin, 1993). This component of the Resiliency Model relies on Lazarus’ and Folkman’s (1984)
definition of coping to explain that coping is viewed as a process that involves continuous
adjustments based on appraisals between the person and the situation. As my research focussed on
parental coping strategies as conceptualised within Lazarus’ and Folkman’s framework, this section
speculates their contribution to family coping patterns and categories for adaptation to managing
CF, and how they are potentially influenced by early surveillance.
Supported by my research findings, parents of children with CF (Tack, et al., 2014; Priddis, et
al., 2010; Zubrycka, 2015), and other chronic conditions (Coffey, 2006; Greening & Stoppelbein,
2007; Tong, et al., 2010), primarily use emotion-focussed coping strategies for coping with
knowledge of their child’s condition. That is, emotion-regulation became a source of coping when it
was understood (i.e., appraised) that CF (i.e., the stressor) could not be removed (Lazarus &
Folkman, 1984). Applied to parents in my research, being able to down-regulate psychological
burden through venting, social comparison, and staying in the present functioned to minimise
personal adversity that helped parents to manage knowing their child has CF.
Downward social comparisons were a prevalent coping strategy parent employed to cope
with their child having CF, but also to cope with their child undergoing early surveillance for lung
178 Parenting Children with Cystic Fibrosis
disease. Parents not only compared their child (and their family) to those who were worse off, but
they also compared their own families to families who were not given the opportunity to track their
child’s health as sensitively as what AREST CF offers. Such appraisals enabled parents to feel a sense
of gratitude for their given situation within the context of wider society, and to cope with their sense
of fear about uncertainty in their child’s morbidity and mortality. Situational appraisals that involved
downward social comparisons functioned as sources of family strength in their capacity to cope with
the demands they faced.
As resources within a family system, individual coping strategies can influence how the
family functions, both in relation to the child’s illness, but also more broadly as a synergistic unit that
works together to harmonise and cultivate itself. The Resiliency Model emphasises how coping
strategies and behaviours of individual members have the capacity to influence family resources of
cohesion, organisation, communication, hardiness, integration and stability. These factors represent
family functioning, essential for the family system’s ability to thrive and for their well-being
(McCubbin & McCubbin, 1993). Moreover, balanced family coping (consisting of integration and
personal growth) has been reported as a predictor of positive health trends in children with CF
(Patterson, et al., 1993). From the point of view of the Resiliency Model, findings from my research
and Patterson et al.’s findings indicate how a family might adapt to their child’s CF, which can
influence a child’s long-term health outcomes.
6.2.5 Social support.
Support from individuals, such as family members and friends, and institutions, such as
medical and community services, can help the way a family copes with an illness stressor (McCubbin
& McCubbin, 1993). The study of social support has received the most attention in literature on
stress associated with family adaptation (Crnic, Greenberg, Ragozin, Robinson & Basham, 1983;
Kazak & Marvin, 1984; Paynter, et al., 2013; Tak & McCubbin, 2002). It has most often been viewed
as a primary buffer or mediator between stress and health breakdown (Horton & Wallander, 2001;
Kazak, 1997). The authors of the Resiliency Model use Cobb’s (1976) early definition of social
support who defined it as information exchange at the interpersonal level that provides; emotional
support (showing individual family members they are loved and cared for); and network support
(showing family members they belong to a network of communication involving mutual support and
understanding). McCubbin and McCubbin (1993) added another form of support to their model that
they felt contribute to a family’s capacity to adapt to their child’s chronic condition; altruistic
support, which is information given in the form of goodwill from others for having given something
of oneself.
179 Parenting Children with Cystic Fibrosis
6.2.5.1 Emotional support.
The primary form of support for parents in my research was emotional support, which
appeared to buffer or mediate fear parents experience around the time of their child’s annual
review and for fear associated with uncertainty of disease progression in their children. As shown in
the previous chapter, emotional support from family and friends helped to regulate personal
burdens, which in turn helped parents to cope with knowledge of their child’s chronic condition. A
question to ask is – are parents whose children undergo early surveillance of CF lung disease in need
of emotional support specifically for their early surveillance experiences? I believe the answer to this
question is yes because of fear associated both with medical procedures and with knowledge of
covert disease progression. It is beneficial to give parents such information about their children but
as HCPs, we need to offer relevant support to manage what it is presented to them. Moreover, the
avoidant coping strategies parents use around the time of annual review may induce depression,
anxiety and/or stress (Sheehan, et al., 2014).
My research findings lay a foundation for understanding specific support that parents need,
and can help to identify appropriate interventions and support strategies. By applying my research
findings to the Resiliency Model, emotional support from family and friends is an important resource
that parents could draw upon to help institute new patterns of family functioning (showing the
bidirectional nature of processes within the Resiliency Model). Additionally, emotional support may
buffer against adverse outcomes from a child’s early surveillance procedures by positively
influencing coping patterns around the time of a child’s annual review.
6.2.5.2 Network support.
Since the discovery that infections associated with CF can be transmitted between patients,
network support has been difficult to offer parents. Changes in clinical practice and community
support have meant that parents and their families are segregated from each other to avoid cross-
infection of children with CF (Bell & Robinson, 2008). There are community-based support programs
offered by Cystic Fibrosis Western Australia that take segregation into consideration (e.g., parent
weekends, dinners and coffee mornings in suburbs and regional areas where only one adult or child
with CF is allowed to attend), and with the introduction of the Internet, there are online support
networks that give parents and their families network-support (e.g., CF Smart and CFMatters
Facebook pages which are both supported by Cystic Fibrosis Western Australia). Additionally,
network support is available to parents from medical and research staff, giving them a source of
coping beyond their immediate networks. However, for parents in my research network-support
from peers, specifically around and regarding early surveillance, is currently lacking. As an important
component for adaptation to chronic illness emphasised by the Resiliency Model, such network-
180 Parenting Children with Cystic Fibrosis
support could enhance parents’ capacity to adapt to their child’s CF in the context of early
surveillance. Moreover, parents in my research highlighted how establishing and maintaining
relationships and networks were important for them to manage their early surveillance experiences.
To facilitate such network support, a formal online support network for AREST CF families where
they could share their stories, ask questions and give advice to each other could form network-
support aimed at developing mutual support and understanding. Mentor training could be offered
to a number of consumer advocates who could moderate the online network so they can be formally
supported throughout the process.
6.2.5.3 Altruistic support.
Altruistic support has unique beneficial elements in the context of my research. Parents
have a sense of altruistic support from knowing they are giving something of themselves and their
child to advancing scientific knowledge about CF to hopefully help future generations of children
born with CF. Whilst the experience of early surveillance is undoubtedly unpleasant, parents are
uniquely supported by the knowledge of their altruistic principles. According to the Resiliency
Model, giving to something that is deemed bigger than oneself can help families to cope with their
child’s chronic condition, and can be a source of family strength that also helps parents to cope.
Therefore, it altruistic support may exist for families coping with CF in the context of early
surveillance.
6.2.6 Coping patterns.
The Resiliency Model indicates relationships exist between family resources and coping
strategies, social support, situational and family-schema appraisal and meaning-making with
generalised coping patterns. The Resiliency Model (McCubbin & McCubbin, 1993) characterises the
family system as a resource-exchange network, whereby coping occurs within the system and as part
of the system. In this way, coping behaviours are the actions for this exchange of resources, resulting
in coping patterns that either facilitate or impede family adaptation to paediatric chronic illness.
Whereas strategies are defined in the Resiliency Model as intentional efforts to reduce or
manage specific illness-related demands, they are grouped into patterns that represent more
generalised ways of responding that transcend different kinds of stressful situations. The authors of
the Resiliency Model identified four broad patterns of coping that facilitate adaptation to illness,
which are inherently either emotion or problem-focussed coping approaches; efforts to reduce or
eliminate stresses and associated hardships, acquisition of additional resources to manage illness
and associated hardships, management of family system tension, and shaping appraisal at both the
situational and schema levels. How these coping patterns are influenced by family resources, social
support, situational and schema appraisal and family meaning-making (as indicated by the Resiliency
181 Parenting Children with Cystic Fibrosis
Model) will be outlined. Further, how coping patterns are influenced by early surveillance through its
contribution to family resources, situational and schema appraisal and family meaning-making will
be outlined.
6.2.6.1 Family resources contribute to family coping patterns.
As a family resource for coping with paediatric chronic disease, gaining the most detailed
information available in the world about a child’s condition gives parents a level of knowledge about
their child’s health that few parents in the world receive. Parents perceive they are gaining as much
information as is conceivably possible about their child’s disease progression. Whilst information-
seeking about the disease itself is a coping strategy, the sensitivity and uniqueness of early
surveillance results appears to give parents an additional element to this particular coping strategy.
Parents perceive a sense of control and empowerment in managing their child’s disease, which,
according to the Resiliency Model, can promote coping with CF more generally.
Additionally, gaining a sense of parenting competence and parental self-esteem by
mastering a child’s home treatment regimen can facilitate coping. Early surveillance results can
confirm these parental constructs when favourable annual review results are received, which serves
to validate parents’ treatment efforts throughout the year. The sense of empowerment, control,
competence and self-esteem are over-arching factors for parents in my research. However,
unfavourable annual review results can undermine these positive experiences.
Whilst parents believe that early surveillance is in the best interests of the child, it can have
short-term, adverse psychological effects on parents. Whilst parents are willing to tolerate these for
the sake of long-term benefits on child health, there is the possibility that these adverse effects may
be experienced on a longer-term basis not be discernable through my research. A longitudinal study
of how individual resources identified in my research are influenced by early surveillance results
annually could be of benefit to understanding any long-term effects.
Lastly, social support is posited in the Resiliency Model to have both a direct effect on coping
patterns, as well as an indirect effect by acting as a family resource. Emotional support and altruistic
support are well-provided for parents in my research, thereby positively contributing to family
resources and coping. However, lack of network support could be detrimental to a family’s capacity
to cope with CF and/or early surveillance, which in turn contribute to adaptation to CF.
Social support has been identified in the literature as a family factor that fosters resiliency
(Benzies & Mychasiuk, 2009). Whether it is instrumental, emotional or practical, support is vital to
families during times of stress and helps maintain good physical and mental health (Black & Ford-
Gilboe, 2004; Walsh, 2003). Therefore, the levels of support identified in the Resiliency Model, and
182 Parenting Children with Cystic Fibrosis
how well they are fulfilled, can influence family coping patterns by providing parents with, or
preventing them from accessing, extraneous sources for individual and family coping.
6.2.6.2 Situational and family schema appraisals and meaning-making contribute to family
coping patterns.
According to the Resiliency Model (McCubbin & McCubbin, 1993), how a family shapes both
their situational and family schema appraisals will contribute to how a family copes with their child’s
condition. Cognitive appraisals play a central role in how one will respond to a stressor, and
therefore what coping mechanisms are activated (Lazarus & Folkman, 1984). This process is
embedded in the Resiliency Model as an outcome for how families adapt to their child’s chronic
condition. In their primary appraisal, parents evaluated their child’s illness as threatening and
harmful. When the condition was considered in this way, negative outcomes were found. However,
social comparisons and early surveillance functioned to change parents’ perceptions of their child’s
CF from a hopeless and uncontrollable situation to one of hope and control, which indicates the
relationship between individual persons and their specific environment (Lazarus & Folkman, 1984).
Consequently, this change appeared to lead to a positive re-evaluation of the child’s condition,
which may lead to subsequent changes in ways of coping. For example, appraisals of a situation as
hopeful and controllable may activate the coping mechanism of sense of mastery for parents, which
is a critical factor in a family’s ability to cope with a crisis situation (McCubbib & McCubbin, 1993).
In this way, the situational and family appraisals of CF in the context of early surveillance,
the resultant creation of hope for their child’s and family’s future, and activated coping strategies
based on situational and family appraisals could operate synergistically to reduce the burden of CF
on families. For example, social comparison is an appraisal that functions as a coping strategy
contributing to a family’s capability to meet the demands it faces. Not only does the appraisal
appear to reduce the psychological burden associated with parenting a child with CF, but the
appraisal appears to activate adaptive coping strategies, further enabling the family to cope with
burden associated with their child’s CF. Moreover, the early surveillance results themselves (and
subsequent treatment) are a mechanism for eliminating, or at least reducing, the perceived severity
of CF. Therefore, these outcomes represent a problem-focussed coping approach for families
managing CF in the context of early surveillance.
Problem-focussed coping aims to eliminate or reduce the source of stress in practical ways,
therefore directly reducing stress (Lazarus & Folkman, 1984). Literature shows that problem-
focussed coping in an array of circumstances is an effective method of decreasing burden associated
with a stressor (Billings & Moos, 1981; Chao, 2012; Moos, Brennan, Fondacaro & Moos, 1990; Nes &
Segerstrom, 2006). This approach has both behavioural and cognitive elements for dealing directly
183 Parenting Children with Cystic Fibrosis
with the stressor, therefore changing the perception of, and relationship with, the stressor. The aim
of AREST CF is to delay or prevent disease progression. By engaging in the AREST CF early
surveillance program, parents are adopting a more problem based coping approach. Parents
primarily utilise emotion-focussed coping strategies for coping with the stress of early surveillance
procedures and processes. Early surveillance provides parents with a practical, problem-focussed
approach to manage stress caused by CF, directly reducing the stress by changing the perception of,
and relationship with, the stressor.
Entering a child into the early surveillance program grants parents a sense of control over CF.
Early surveillance participation changes parents’ perceptions of CF through the early, proactive
management of the disease afforded by early detection. Parents felt as though they had taken as
much control as they possibly could over CF by entering their child into early surveillance; which
parents attribute to the uniqueness of this type of CF management. Access to the information from
early surveillance allows parents to cope more generally with CF. Therefore, a sense of mastery and
acquiring knowledge and skills makes parents feel as though they are taking control of the situation
that has been presented to them – affording them a problem-focussed coping mechanism to coping
with CF. By feeling a sense of control and a sense of mastery, parents’ self-esteem is supported.
Therefore, the empowerment and perceived control (i.e., taking control) parents experience from
acquiring knowledge (i.e., information-seeking) about their child’s condition, as well as from their
understanding of sensitivity and uniqueness of early surveillance, promotes coping with CF more
generally. It is conceivable that parents whose children undergo early surveillance may be less
daunted by, more optimistic about, and perceived to be more in control of their child’s CF than
parents whose children do not undergo early surveillance, which may have implications for parents’
attitudes and values around treatment adherence, as well parental identity and parental mental
health and quality of life more generally.
Lastly, the constructs of situational and family schema appraisals and meaning-making
emphasised in the Resiliency Model (McCubbin & McCubbin, 1993) interact directly with what is
labelled as situation and family schema appraisal coping. That is, what a family values and prioritises
(i.e., family schema appraisal), and what they think of their situation (i.e., situational appraisal) and
the meaning they attach to it (i.e., meaning-making), can be a generalised coping pattern for
adapting to a child’s chronic condition. Gratitude, perceived control and power from situational
appraisal, along with the meaning of hope created from both levels of appraisal, emerged as coping
mechanisms through which parents were able to feel comfort and content with their child’s CF in the
context of early surveillance; maybe because of the context of early surveillance.
184 Parenting Children with Cystic Fibrosis
With appraisals identified at the individual level, and their influence on coping discerned,
how these appraisals might function synergistically or antagonistically to produce family adaptation
could be explored in the future. As the Resiliency Model emphasises family-level appraisal as a
determining factor of whether other illness dimensions become problematic, identifying individual-
level factors is an important part of creating a comprehensive understanding of how families adapt
to paediatric chronic illness. With little research conducted on the family-level appraisal component
of the Resiliency Model, the framework could be used to distinguish family appraisals that represent
the unique experience of early surveillance and how those factors might facilitate or impede family
adaptation to CF. How the unique experience of early surveillance might compare to other families
experiencing paediatric chronic illness can also be explored to determine commonalities and points
of difference that can be viewed in the broader context of paediatric chronic conditions.
6.2.7 The family adaptation process: Bonadaptation and maladaptation.
The final outcome within the Resiliency Model is family adaptation, where families engage in
direct response to the excessive demands of an illness and depleted resources, and realise that
systemic changes are required to restore functional stability and improve family satisfaction within
their given situation (McCubbin & McCubbin, 1993). The Resiliency Model highlights how coping
strategies play a critical role in adaptation, which is the point where my thesis ends and future
research must take over. My thesis has highlighted what parents must cope with in relation to their
child’s CF in the context of early surveillance. My thesis has further highlighted numerous ways that
parents strive to cope with their child’s CF, and particularly in the context of early surveillance, and
how early surveillance can facilitate or impede coping, and how engagement in surveillance itself,
can be used as a coping mechanism.
6.3 Summary and conclusion
Using the Resiliency Model as a guiding framework, this chapter has shown how families
might adapt to paediatric chronic illness through a series of inter-related processes and resources.
The first change required for family adaptation is that newly instituted patterns of family functioning
must be engaged. Parents in my research described how they normalised CF and its treatment and
redefined future expectations, which helped them to form new patterns of practical and existential
family functioning.
Appraisals at both levels and meaning-making were positively influenced by participating in
early surveillance, whereas early surveillance participation could have either positive or negative
effects on family resources such as sense of mastery and self-esteem, depending on what annual
review surveillance results yielded. Therefore, the Resiliency Model aptly highlights how parents’
185 Parenting Children with Cystic Fibrosis
adaptation to their child’s CF can be influenced by early surveillance, and can therefore be used a
guiding framework to develop and implement tailored interventions.
For example, application of the Resiliency Model to my research findings highlights that
parents generally normalise CF into the family and establish patterns of family functioning that
incorporate the treatment regimen. Additionally, parents develop expectations that are congruent
with their newly instituted patterns of family functioning because of the hope they attribute to their
family meaning. Applied to the Resiliency Model, my findings highlight that hope for a child’s future
establishes a link between newly instituted patterns of family functioning and their expectations of
those new patterns. The combination of compatible patterns (or actions) and expectations (or
thoughts) contribute to effective family coping with paediatric chronic illness. The process from
establishing patterns of family functioning to effective family coping demonstrates how the
Resiliency Model can be used in clinical practice as a framework to assess how families might adapt
to paediatric chronic disease in the context of early surveillance for disease progression. Due to the
uniqueness of early surveillance for CF lung disease, how a program such as that run by AREST CF
functions to facilitate coping has not previously been reported. Indeed, it would seem that early
surveillance is a primary mechanism for coping with CF.
Subsequently, it is possible that parents whose children undergo early surveillance for CF
lung disease have more coping mechanisms than parents whose children do not undergo early
surveillance for CF lung disease. Therefore, parents in my research may be more resilient for future
burden of CF because of the additional coping mechanisms parents construct from their early
surveillance involvement. However, the possibility that these parents are more vulnerable to future
of burden of CF must be considered because of the additional stressors of early surveillance, and
their effects on parents, identified as part of my research. Moreover, problem-focussed coping is
generally more effective than emotion-focussed coping, which may mean that parents cope
effectively with their child’s CF but not so well with their child’s early surveillance.
In conclusion, the Resiliency Model attempts to explain how families adapt to paediatric
chronic illness by isolating individual, family and community properties and processes that interact
and shape the course of family behaviour over time. My thesis has been able to isolate individual
factors from parents’ perspective that are posited within the Resiliency Model to contribute to
family adaptation to paediatric chronic illness. Additionally, considerations for how constructions of
experience and attributions of meaning may contribute to coping with lived experiences were made.
My thesis has demonstrated use of the Resiliency Model as a framework that would assist in
determining how families might respond to CF within the context of early surveillance, which may
lead to family resiliency and adaptation. The model presented within my thesis characterises how
186 Parenting Children with Cystic Fibrosis
families might respond to CF within the context of early surveillance so that targeted interventions
can be tailored to family needs that build individual and family strength. The model can therefore be
aptly used as a framework to assess individual and family-level factors that may function as
predictors of family adaptation in the context of early surveillance for CF lung disease. The factors
identified in my research that are outlined in the model presented in the thesis can be tested
longitudinally through mixed-effects logistical regression analyses to determine indicators of health
vulnerability and resilience. Subsequently, intervention and support can target specific individual
and family-level factors that may reduce risk of maladaptation, as well as increase adaptation and
resilience.
This chapter has provided a stepping stone from my research questions about parental
perspectives to the more holistic biopsychosocial model of family existence, health and resilience.
The Resiliency Model of Stress, Adjustment and Adaptation distinguishes psychological processes,
and coping strategies and styles that likely contribute to coping with a paediatric chronic condition.
Using this particular theoretical framework to interpret and extrapolate findings has allowed
application of my research findings beyond their current context that will hopefully drive further
exploration and analyses.
Lastly, the relationships identified in the Resiliency Model are bi-directional, meaning that
each component within the model has a reciprocal nature with its associated components.
Therefore, adaptation to CF is not static, nor are the elements of family life that contribute to it. As
families move through their developmental trajectory and as illness-related crises demand attention,
parents need to reconsider and adjust to changes for their families to continue adaptation to a
child’s progressive condition. The implications of my research findings and their theoretical and
practical application to best support parents during diagnosis and early surveillance for CF lung
disease are reviewed in further detail in the next chapter.
187 Parenting Children with Cystic Fibrosis
CHAPTER 7
Summary and conclusions: Key research findings, implications, strengths, limitations and future directions
In this chapter, key findings are summarised to give an account of, and the reasons for,
parents’ experiences of parenting children with CF in the context of early surveillance.
Findings and interpretations from the data are presented and positioned within the
context of the main research aims and questions. The significant aspects relating to
parents’ experiences of early surveillance are also summarised along with how parents
cope, how they construct their experience and what meaning they attribute to early
surveillance. Implications of this research for service delivery pertinent to clinical care as
well as implications for family theory, policy and practice are discussed. To conclude the
chapter, strengths, contributions and limitations of this research are reviewed, avenues
for future research outlined, and lastly, post-reflections of conducting the research are
described.
188 Parenting Children with Cystic Fibrosis
Hope is the thing with feathers, that perches in the soul, and sings the tune without the word,
and never stops at all…Emily Dickinson
7.1 Key findings that apply to research aims and questions
The primary aim of this research was to explore the lived experience of parenting children
with CF in the context of early surveillance. An additional aim was to explore how parents cope with
the lived experience of parenting. Moreover, my research aimed to explore how parents construct
their experience and attribute meaning to early surveillance for CF lung disease. The aims of the
research were achieved using in-depth semi-structured interview data to explore experiences of CF
diagnosis, treatment management and early surveillance. Examining parents’ experiences,
constructions and attributions of meaning in the context of current conceptualisations of adaptation
to paediatric chronic conditions also facilitated achievement of the research aims. The exploratory
nature of the research was essential given the current focus on early intervention for chronic
conditions; as a response to recent national and international clinical and political agendas on early
intervention (Jenkins, 2005; Wise, da Silva, Webster & Sanson, 2006), and due to the dearth of
research about the lived experience of parenting a child with CF in the context of early surveillance
from the perspective of parents themselves. The key findings from this research are summarised in
the next section and reviewed in context of the research questions.
7.2 The lived experience of parenting a child with CF in the context of early surveillance:
Implications for policy and practice
Research questions one and two:
1. What is the lived experience of parenting children with CF in the context of early
surveillance?
2. What are parents’ experiences of early surveillance?
Essentially, the aim of all research is to contribute to the knowledge base about an area of
interest (Mays & Pope, 2000). In my research, parents’ lived experiences and personal accounts both
informed and extended existing knowledge about the experience of parenting a child with CF. A
salient finding was that common experiences and thoughts were shared by parents, regardless of
their gender or the age of their child. As such, findings and interpretations relating to research
questions one and two (as outlined above) were presented collectively. Where relevant, findings
unique to mothers or fathers, or parents of children younger or older than 3 years of age were
presented separately. A summary of the key findings relevant to research questions one and two are
reviewed in the next section and their contribution to contemporary understandings of parenting a
189 Parenting Children with Cystic Fibrosis
child with CF and in particular, are discussed in relation to implications they have for future policy
and practice.
7.2.1 Policy and practice implications.
The current national and international political and clinical agendas for early intervention
(Jenkins, 2005; Wise, da Silva, Webster & Sanson, 2006) prompted my research to explore the lived
experience of parenting a child within the context of early surveillance for CF lung disease, and to
investigate parents’ experiences of the early surveillance process itself. The unique context of early
surveillance for CF lung disease gave me the opportunity of exploring the lived experience of
parenting children who undergo early and aggressive treatment for CF lung disease.
As previously established, the biomedical view of chronic disease has largely constructed
early diagnosis and intervention as a modern conception for prevention of disease progression
rather than from a therapeutic perspective. My research found that while personal belief
orientations and clinically contextual factors function to advocate early diagnosis and surveillance to
parents, it was also evident that psychological and social aspects of the experience were of equal or
greater significance to the meaningful understandings parents constructed about their experience of
parenting a child within the context of early surveillance. The findings of my research highlight a
need to evaluate the way in which diagnosis and early surveillance are constructed and normalised,
both in terms of the practical and experiential, so that a holistic view of CF care can be used as a
basis to support parents through such a unique process.
By its very nature, normalisation of paediatric chronic illness and its treatment into the
family context may buffer and protect its members against its existence as well as against future
burdens of the condition. Moreover, though life is disrupted at diagnosis, early diagnosis resulting
from NBS allows normalisation to occur very early in the child’s life (Cederborg, et al., 2011).
Therefore, normalisation of the condition and its treatment, both as a process and an outcome
should be an endeavour of the family, clinicians and other HCPs working with these families. The
more established and routine a family can become, the more accessible normalisation, and its
benefits, will be. The inextricable link identified in my research between CF and early surveillance
meant that parents’ normalisation of CF was referenced in relation to early surveillance, giving
parents an additional reference point that functioned to create hope and control in a situation that
was initially constructed as hopeless and uncontrollable. Therefore, understanding how parents
appraise circumstances presented to them, and what factors contribute, becomes a critical
component of normalisation when evaluating how parents construct their experiences and attribute
meanings.
190 Parenting Children with Cystic Fibrosis
The design of the AREST CF early surveillance program focusses on physical aspects of a
child’s health and as such, parents’ main motivation for becoming involved in the program included
to address their child’s current health condition, and as a preventative measure to preserve their
child’s health. Among the features identified as important to parents’ experiences included
knowledge of a child’s current health status as well as any suggested treatment as a result of the
surveillance. Therefore, parents’ accounts of their child’s early surveillance provide support for the
perceived importance of early intervention and preventive action against disease progression from
both a parental and clinical perspective.
Though loss of connection between parents of children with CF due to recent
understandings of cross-infection is substantiated (Bell & Robinson, 2008), parents desire
connection with other parents under the same unique circumstance (Duffy, 2011; Eatough, et al.,
2013; Kerr, et al., 2004; Shields, et al., 2008). Importance of this aspect of early surveillance does not
appear fully appreciated, and therefore alternative considerations have not been fully explored. This
particular finding emphasises that connection with other parents and families could be an essential
component of parental adaptation to their child’s CF within the unique context of early surveillance
(Barcroft, 2015). Findings also indicated that parents respond positively to a well-structured and
comprehensively yet simplistically explained program with experienced and trustworthy staff that
relate and communicate well.
Development of rapport with parents early in the therapeutic relationship is of paramount
importance to clinicians because of the ongoing medical care required for CF patients. Emphasising
importance of the therapeutic relationship, Swallow & Jacoby (2001), Salgado, et al. (2011) and
Gjengedal et al. (2003) reported how this type of relationship provided a consistent source of
support for parents. Spiers, et al. (2011) suggested that such relationships are integral for parents’
confidence to administer home treatment, and Bull & Grogan (2010) asserted that it was vital that
parents felt confident in the health professional they were relinquishing their child to. Given the
importance of the therapeutic relationship for CF patients, clinicians and researchers introducing
early surveillance need to be considerate of parents’ experience during this time. For example,
parents were more likely to feel confident about their child’s involvement when they felt a sense of
competence and trust in the hospital staff.
Notwithstanding these factors, the most salient finding to emerge from parents’ experiences
of the early surveillance program was that knowledge acquisition about a child’s health status was
critically important to parents’ sustained involvement. In particular, a sense of control over disease
progression and sense of hope for the child’s future contributed to parents’ positive experiences of
the early surveillance program. Whilst there are undoubtedly adverse experiences, reactions and
191 Parenting Children with Cystic Fibrosis
outcomes that require consideration for intervention and support strategies, parents’ perceptions of
themselves were generally positively influenced by their involvement. This was particularly
evidenced by their perceived parenting competence being influenced by the level of self-efficacy for
achieving their child’s home treatment regimen and orientation towards an internal locus of control
for their child’s health, which contributed to increased parenting self-esteem. Moreover, feelings of
altruism and gratitude for the opportunity to participate in early surveillance positively influenced
parents’ lives.
However, receiving knowledge about a child’s covert disease progression as part of
involvement in early surveillance for CF lung disease appears double-edged. That is, parents perceive
both control over disease progression and also hope for their child’s future, yet they can become
acutely attuned to how their child’s condition is likely to progress, which can result in fear (Glasscoe
& Smith, 2011). Dimensions of fear in the context of early surveillance for CF include fear of losing a
child to CF, fear of a child getting sick from disease severity, and fear of knowing a child is getting
sick through knowledge of early surveillance results. These dimensions of fear are inextricably linked
to, and affected by, each other. For example, fear that a child will die young is fear of disease
progression, and so because the child may die of disease progression, the disease, too, becomes an
object of fear. Then, because early surveillance provides knowledge of disease progression, which
for parents can be viewed as an indication of the child’s life expectancy early surveillance, too,
becomes an object of fear.
Parents want knowledge of their child’s often covert disease progression so, if present, it can
be treated as soon as possible; but they are also fearful of it because of what it means for their child
and their family. Therefore, it appears that parents simultaneously fear and desire knowledge of
their child’s covert disease progression. The early surveillance program provides parents with
detailed information about their child’s disease progression, but parents have not been provided
with tools for how to manage such information. Furthermore, is the issue of perceived accountability
for child health by parents as primary carers and receiving detailed information about their child’s
disease progression. Given the findings of my research, there is an opportunity to support parents
through such experiences by connecting parents with other families in the AREST CF program so
personal experiences can be shared in such a way that mutual understandings are created. Such
support could enable parents to develop realistic expectations about the early surveillance
experience, as well as about their parental role in the care for their child.
The adverse experiences, reactions and outcomes that occurred of parents need to be
considered as likely influencing parental mental health. Among the challenges experienced by
parents were; feelings of self-judgement and blame if their child’s annual review results showed
192 Parenting Children with Cystic Fibrosis
some disease progression; relinquishment of their child for medical procedures, and cognitive
dissonance around the decision to enrol their child in medical research. Becoming familiar with the
processes and procedures, and with the research and medical staff appeared to mitigate adverse
responses. These findings bring attention to the issues that parents encounter and that hospital staff
must attend to so that parents can be better supported in their quest to keep their children healthy.
A reciprocal opportunity exists for future research to consult with medical and research staff about
their experience of running such a program, as well as their experiences of parents and children
attending. This type of research may identify where staff perceptions are the same and different to
parents, as well as any assumptions or anxieties for staff.
Moreover, my research has identified a need to assess whether parental anxiety is prevalent
among parents of children undergoing early surveillance of this nature in CF, and determine what
resiliency techniques might mitigate it, if it indeed needs addressing. Based on my findings and
reports in the paediatric medical literature (Bearden, et al., 2012; Caes, et al., 2014; Harper, et al.,
2013; Karlsson, Englund, Enskär & Rydström 2014; Kwan, Chiu, Gan & Chan 2016; Scholten, 2013),
intervention for fear and anxiety, lack of control and helplessness, cognitive dissonance, or damage
to parent identity may be necessary to support parents throughout their child’s annual early
surveillance procedures. For example, narrative psychotherapy is a forum where parents could
express their fears and anxieties in a safe and supportive environment, where concerns can be
validated and, at the same time, demystified and moderated. As an in-the-moment strategy, parents
could use mindfulness techniques to help regulate either anxiety associated with relinquishment or
cognitive dissonance during their child’s medical procedures. Mindfulness strategies have
demonstrated ability to reduce stress (Brown, Warren & Richard, 2003) and anxiety (Evans, et al.,
2008; Hofmann, Sawyer, Witt & Oh, 2010), and could replace the distraction technique of coping
with relinquishing a child for medical procedures. Mindfulness-based education could form part of
the narrative psychotherapy. The social work or psychological annual assessment could include
provision for mindfulness education and an assessment for anxiety using a brief screening tool such
as The Generalised Anxiety Disorder Questionnaire (GAD-7) (Spitzer, et al., 2006). The GAD-7 has
recently been recommended for use with parents of children with CF by The Cystic Fibrosis
Foundation and the European Cystic Fibrosis Society Guidelines Committee on Mental Health
(Quittner, et al., 2015). Additionally, the Reaction to Diagnosis interview (Pianta & Marvin, 1993) is a
brief measure of grief associated with diagnosis that the social worker or psychologist could
administer to parents to identify early, potential psychopathology.
Recently, substantial attention has been given to the role of translational research in public
health systems and the provision of health care services (Oborn, et al., 2010; Ogilvie, et al., 2009).
193 Parenting Children with Cystic Fibrosis
This view emerged from recognition that health systems across the world often did not reach their
full potential in the provision of health services (World Health Organization, 2004), and that health
knowledge did not always translate into practice (Woolf, 2008). Findings from my research
demonstrate a gap exists between the translation of the current lived experience and
understandings of early diagnosis and early surveillance held by parents into clinical practices among
the treating team. For example, improving or enhancing family resiliency appears an appropriate
and sustainable approach to caring for children undergoing early surveillance within the wider family
context. Benefits of such as an approach should be systemic and reciprocal; benefits for child and
the family system, and benefits for the child because of the care for the family system. A benefit
might be that experiential effects of early surveillance from a psychological perspective are more
clearly defined, which could result in targeted support for parents and their families as they embark
on a unique experience that has little specific psychological support currently built into it.
In review, my research highlights the importance of continual conversation that needs to be
had between the treating team and the parents about their experiences of parenting within the
context of early surveillance for CF lung disease to ensure knowledge is better translated into policy
and practice as real-world application. To sustain such a process, parents first need to feel
appreciated, and that what they have to say is important and valued. Although recent clinical
guidelines advocate for early intervention, parents in my research often expressed that their
experiences with clinical practices and interactions with clinical staff were not always fully
understood. As such, my research indicates a disconnect between clinical practice and the actual
lived experience of early surveillance. My research highlights the substantial role of qualitative
research in exploring the lived experience of parenting a child with CF undergoing early surveillance
though moreover, underpinning factors have been identified for development of a framework that
would serve not only to inform clinical practice and policy from a holistic perspective, but to critically
assess how knowledge of the lived experience of parenting a child undergoing early surveillance for
CF lung disease is translated into practice.
7.3 Parents’ constructions and attributions of meaning to early surveillance
Research question three:
3. How do parents construct their experience and attribute meanings to early surveillance for
CF lung disease?
A salient finding of my research was evidence that constructions of experience and
attributions of meaning were foremost primed by the wider societal discourses used to promote
early intervention in paediatric chronic disease. Parents’ constructions of early surveillance were
194 Parenting Children with Cystic Fibrosis
particularly influenced by biomedical discourses prevalent in wider society that emphasised an early
intervention understanding of paediatric chronic disease. Findings also revealed that the notion of
early intervention was highly familiar to parents and was found to influence both how they thought
about adherence to their child’s treatment regimen as well as how they constructed meaningful
understandings about the experience of early surveillance. In particular, proactive and preventive
treatment was of paramount importance to parents given the chronic nature of the condition. For
example, parents were keen to know their children were being treated as early as possible. This
finding reinforced how discourses within social contexts can influence how parents related to their
own personal experiences of CF in the context of early surveillance.
Moreover, parents revealed that making social comparisons with other families encountered
within the hospital environment as well as with families whose child has CF, but who does not
undergo early surveillance for CF lung disease, was a salient element of constructing meaning about
their own experiences of CF. Making comparisons with others enabled parents to retain both a
positive self-perception and perception of their family’s present and future (Moola, 2012; Tanner,
2007). This tendency allowed parents to reflect on their fears, hopes and plans for the future (Giles,
McIlrath, Mulac & McCann, 2010). Furthermore, making downward comparisons appeared to
sustain parents’ motivation to fulfil personally meaningful goals such as mastery of the treatment
regimen and normalisation of the condition into family life, a finding that was consistent with
previous research (Goldberg, et al., 1990; Heaton, 2014; Hughes & Beer, 2012). Additionally, the
data suggest that the effects of making downward comparisons may not only be relevant for
parenting a child with CF but also for the early surveillance experience, which resulted in hope for
the future and gratitude for the present, a finding that would benefit from further research.
Therefore, the current findings both support and extend existing social comparison literature
(Festinger, 1964; Kwan, et al., 2003; Moola, 2012; Tanner, 2007).
My research supports the notion that meaningful understandings of early surveillance are
more than the clinical outcome. Rather, with the influence of knowledge on constructing meaning
about early surveillance, an opportunity presents itself to understand what becomes of importance
for parents who enrol their children in early surveillance for disease progression. With parents’ lived
experiences reflecting personal outcomes of hope, gratitude, control and power, this finding
suggests that early intervention initiatives need to embrace a holistic awareness of all aspects that
influence parents’ experience of the child’s condition. It became evident that future policy decision
making and models of practice should be encouraged to focus on the advocacy parental experience
in a way that more closely aligns with their lived experiences. An area identified in my research
where policy makers might translate knowledge into practice includes growing acknowledgement of
195 Parenting Children with Cystic Fibrosis
a link between self-efficacy, locus of control and self-esteem (Judge, Erez, Bono & Thoresen, 2002;
Pruessner, et al., 2005; Wang, Zhang & Jackson, 2013; Wilson, et al., 2013), and how that link can be
applied to parents’ well-being in the context of early surveillance. For example, a resiliency approach
to holistic care could be used to educate parents (and clinicians) about how taking ownership over
their child’s care will contribute to confidence in their redefined parental roles. Broader strategies
may include reviewing the way early surveillance is introduced to parents, including timing, content
and modality of information, or development and implementation of a standardised protocol for its
introduction.
Moreover, my findings suggest that future policy and practice needs to better acknowledge
and critically evaluate relationships between experiential, practical and psychological factors
associated with early surveillance in shaping parents’ thoughts and emotions around their child’s
and their family’s future, their perceptions of themselves as parents, and their hopes, dreams and
fears. A critical examination of the relationship between such factors would generate further
understanding of both beneficial and adverse influences of early surveillance on parents’
constructions of their experiences and attributions of meaning to them. Much like the sense of
moral and social obligation held by the CIHR to ensure that outcomes of research proceed back to
individuals who could benefit (Canadian Institutes for Health research, 2004), an essential
component of the investigation would not just involve critically thinking about parental experience
of clinical structures and practices. Rather, it would extend to recognise that those in positions of
power who are in support of early surveillance need to consider parental consequences of such
decisions and actions, which are now recognised as outcomes of my research. As demonstrated by
my research, lack of both standard protocol and therapeutic support denies substantial adverse
influence of early surveillance on parents, as well as negates potential misconceptions about
research and clinical care that could affect autonomous decision-making. Moreover, lack of
education about beneficial influence of early surveillance to parents also denies them full potential
psychological and experiential benefits.
7.4 Coping with parenting children with CF in the context of early surveillance: Implications for
theory, policy and practice
Research question four:
4. How do parents cope with the lived experience of parenting in the context of early
surveillance?
Further to exploring the lived experience of parenting a child within the context of early
surveillance for CF lung disease was to explore how parents cope with their experiences. Again, the
196 Parenting Children with Cystic Fibrosis
unique context gave me the opportunity of exploring how parents cope with the lived experience of
parenting within the context of early and aggressive treatment for CF lung disease. The findings of
my research have a number of important implications for future theory, policy and practice.
7.4.1 Theoretical implications.
My research has described and demonstrated that there are strong indications that parents
generally cope well and adapt functionally to their child’s CF diagnosis and treatment within the
context of an early surveillance regimen. Parents’ perceptions of hope, perceived control and power
means that a resiliency perspective can further foster existing hope by inviting the family unit to
recognise and call on their inherent strengths and coping abilities. Moreover, my research supports
contemporary conceptualisations of resilience models for coping with paediatric chronic conditions,
which if utilised in clinical practice, can encourage HCPs working with families to recognise resiliency
and the inherent strengths within the family unit. If understood by HCPs, resiliency characteristics
and inherent family strengths could become targets for strengths-based intervention. Lastly, a
resiliency perspective would provide information needed to develop preventive interventions for
families. For example, social comparison has been identified in my research as an inherent strength
that reduces the intensity of burden and demands that families face, and activates adaptive coping
strategies. Such information could be used in the development and implementation of preventative
interventions.
Family resiliency theory highlights the complex but meaningful role that family properties,
behaviours and capabilities play in buffering the impact of stressful life events, as well as facilitating
the family’s recovery in the face of family crisis, such as diagnosis of a paediatric chronic condition.
From a salutogenic point of view, the family resiliency framework can be used to discover and build
upon parents’ and family’s’ existing strengths and resources, which makes it particularly well-suited
to family-focussed, resilience-oriented work with families. A crucial initial step to such work is to
identify existing and potential skills, attitudes and other resources that may enhance the family’s
overall growth and response to adverse circumstances. Such an approach runs counter to the
traditional deficit-based models of assessment and intervention that have dominated training and
practice in the helping professions. Traditional family assessment is based on the medical model
assumptions and strategies aimed at identifying and treating family pathology and dysfunction
(Goldenberg & Goldenberg, 2004). Using a resilience framework can help practitioners identify
aspects of family resilience that may go underutilised by the deficit-based model traditionally used in
clinical care.
My research has shown that parents use emotion-focussed and avoidant coping for
managing early surveillance, yet they use early surveillance as an instrumental and problem-
197 Parenting Children with Cystic Fibrosis
focussed coping strategy for managing CF. Literature supports negative outcomes for emotion-
focussed and avoidant coping styles including low emotional and social quality of life (McHugh &
Otto, 2012), as well as depression, anxiety and stress (Sheehan, et al., 2014), suggesting that coping
with early surveillance may be detrimental to parents’ well-being. Additionally, is the concern of
chronic sorrow around the time of the annual early surveillance review. However, instrumental and
problem-focussed coping has been associated with high emotional and social quality of life (McHugh
& Otto, 2012). Therefore, early surveillance may function to decrease social and emotional quality of
life, as well as increase depression, anxiety and stress, and potentially trigger chronic sorrow around
the time of annual review. However, early surveillance may function to increase emotional and
social quality of life on a more long-term basis.
The potential for decreased social and emotional quality of life, along with depression,
anxiety and chronic sorrow around the time of annual review as a function of increased social and
emotional quality of life on a longer-term basis provides evidence that early surveillance is in the
best interest of the child and their parents, but that it is a psychologically distressing experience for
the family. Parents who behaviourally or psychologically disengage prior to annual review and/or
cognitively distract themselves around the time of annual review may be at increased risk of low
social quality of life during this time. This could be critical for parents’ coping capacity because my
research has shown that social support is integral to managing the early surveillance experience.
Furthermore, that short-term negative experiences (i.e., cognitive dissonance, fear for their child’s
safety) appear a compromise for long-term positive outcomes (perceived control power, hope,
gratitude, and altruism) may counteract the negativity experienced at annual review. Essentially, any
psychological adversity experienced by parents as a result of their child’s early surveillance exposure
appears to be short-term adverse individual effects that are accounted for by long-term positive
child health outcomes and beneficial psychological parental outcomes. However, there is still the
concern of triggering chronic sorrow that by definition, is continuous and recurring.
My research reinforced prior literature that has emphasised the importance of social
support for parents coping with paediatric chronic disease (Greening & Stoppelbein, 2007; Hoekstra-
Weebers, et al., 2001; Horton & Wallander, 2001; Wiedebusch, et al., 2010). Unique to my research
was the finding that social support contributed to multiple elements of the CF experience within the
context of early surveillance. Support from family, friends and both the medical and research teams
contributed to the way parents; managed their child’s diagnosis and treatment regimen, normalised
CF and the treatment regimen into family life, coped with their fear associated both with uncertainty
of CF prognosis and with their child’s early surveillance procedures. The data also support previous
research that has described the importance of social support for coping with medical procedures
198 Parenting Children with Cystic Fibrosis
(Marsac, Donlon, Winston & Kassam-Adams, 2013; Peterson, 2013). Therefore, social support as a
primary coping resource for fear associated both with early surveillance procedures and with fear of
disease progression highlights the importance and value parents place on establishing and
maintaining relationships and networks. These findings compliment theoretical advocacy in the
literature arguing for the importance of social support in adaptation to paediatric chronic disease
(McCubbin & McCubbin, 1993; Walsh, 2003). Lastly, findings support previous research regarding
the importance of recognising and addressing such existential needs in holistic approaches to clinical
care for children with chronic conditions (Bekenkamp, Groothof, Bloemers & Tomic, 2014; Shilling,
et al., 2013).
The findings of my research also build upon prominent theories about the importance of
developing and maintaining a perceived competent parental role as individuals transition, and
adjust, to parenthood (Cast, 2004; McBride & Toller, 2011; O'Connor & Barrera, 2014). In my
research, parents reported that perceiving themselves as a competent parent within the context of
their child’s condition was essential in exercising meaningful practices that contribute to perceptions
of themselves as parents. Moreover, parenting practices and perceptions were found to be
important markers for parents in understanding their own parenting experience. Despite challenges,
parents were generally able to regulate their psychological states successfully as indicated by
research into other paediatric chronic conditions (Clark, et al., 2014; Dardas & Ahmad, 2015; Knafl,
et al., 2013; Ruiz-Robledillo, De Andrés-García, Pérez-Blasco, González-Bono & Moya-Albiol, 2014).
Parents achieved psychological regulation (including emotion) through maintenance of a positive
attitude, primarily by way of accepting and assimilating CF and its treatment into family life, as well
as redefining parent identity. Parents were able to maintain hope (Barlow & Ellard, 2006; Faso, et al.,
2013; Horton & Wallander, 2001; Kashdan, et al., 2002; Lloyd & Hastings, 2009; Ogston, Mackintosh
& Myers, 2011; Petersen & Wilkinson, 2015; Samson, et al., 2009; Wong & Heriot, 2008), and
negotiate the dynamics of gains versus adversities to achieve those gains. These findings provide
support for the assumption that families with positive meaning and appraisal are likely to adapt to
paediatric chronic disease, as well as be resilient to future burden of the condition (Black & Lobo,
2008; Lazarus & Folkman, 1984; McCubbin & McCubbin, 1996).
Lastly, normalisation of CF into the family setting and positive meaning-making about the
condition were of primary importance to the CF experience within the context of early surveillance,
which supports the Resiliency Model’s emphasis on them as important processes for adaptation.
Moreover, my research has highlighted how early surveillance can positively and negatively
influence these processes. Therefore, these findings suggest that families who are able to draw
meaningful inferences about CF and early surveillance, and establish newly instituted patterns of
199 Parenting Children with Cystic Fibrosis
family functioning are likely to adapt to paediatric chronic disease as well as be resilient to future
burden of the condition (Black & Lobo, 2008; McCubbin & McCubbin, 1996). Furthermore, family
processes and outcomes of adapting to CF should be viewed within the context of early surveillance
because they are part of the same experience.
7.4.2 Policy and practice implications.
Policy and practice implications of my findings requires thinking about, and implementing,
support strategies that promote and/or enhance adaptation to paediatric chronic disease within the
unique context of early surveillance for covert disease progression. For example, resiliency-building
strategies could be implemented as part of the annual review process when the social worker or
psychologist within the multidisciplinary team offers his/her support to the family. Such strategies
could inform a support program designed to build capacity and develop resources for parents’
coping with adversity. The offer of support could include elements of education, as well as referral
to resiliency-building community programs. Implementing support strategies through policy and
practice, as well as thinking beyond clinical practice, is of primary importance so that parents can
nurture their families as competent and supported parents.
Firstly, family resiliency substantially influences coping with, and adjustment to, life stressors
and events such as paediatric chronic illness for all family members (Giallo & Gavidia-Payne, 2006;
Knafl, et al., 2013; Van Schoors, et al, 2015). Early surveillance of covert disease has been shown to
positively and negatively influence processes that make up family resiliency, that is, positive
behavioural patterns and functional competence. Therefore, building and maintaining family
resiliency should be a focus for paediatric clinical teams for families in their care because reinforcing
family resilience may enhance families’ coping and adaptation abilities/capabilities. Additionally,
despite hardships and stresses of early surveillance specifically, and CF more generally, given the
findings of my research, a strengths-based approach to managing families appears an appropriate
model of support.
Implementing family resiliency frameworks that build program-specific experiences into
existing support programs for parents of chronically ill children have shown promising outcomes (eg.
Kieckhefer, et al., 2013; Mullins, et al., 2015). Accordingly, developing a resiliency program specific
for early surveillance of covert disease could utilise elements of the early surveillance experience
identified in my research that are known to support development of resiliency, for example, self-
efficacy, locus of control, and adaptive coping (Kilic, Dorstyn & Guiver, 2013; Skinner, Pitzer & Steele,
2013; Wang, et al., 2013). Moreover, based on theoretical advocacy and support from my research,
as well as others (Mitmansgruber, et al., 2015; Horton & Wallander, 2001), a number of strategies
could be considered within a resiliency framework as being influenced by the early surveillance
200 Parenting Children with Cystic Fibrosis
process that are known to contribute to adaptation and resiliency when faced with stressful events.
Firstly, strategies towards building family resiliency could: cultivate strong support networks both
within and outside the family to help find understanding, practical advice and meaning (i.e.,
resources for social support); set goals that include hope for the future and motivation for the family
to stick together (i.e., sense of coherence); learn as much about the condition, treatment and child
health status as possible and educate family members about therapies and ongoing research (i.e.,
individual resource that contributes to family adaptation); and hold a belief in a greater entity than
thyself (i.e., altruism). The adapted Resiliency Model presented in the previous chapter is a
framework that could be used to guide development of a resiliency program. The program could
focus on developing those family characteristics of resilience that I demonstrated as contributing to
adaptation to paediatric chronic disease in the context of an early surveillance program.
Secondly, emotion and problem-focussed coping strategies I have identified assists our
understanding of the role family resources play in parents’ management of their child’s CF, both
practically and experientially. As family resources, these coping strategies contribute to a family’s
coping patterns. Problem-focussed coping strategies are not as commonly reported in the literature
as emotion-focussed coping strategies are for parents of children with CF, as they have not
traditionally been afforded such coping mechanisms. Due to the uniqueness of early surveillance,
how a program such as AREST CF functions to facilitate coping has not previously been reported.
Through access to, and execution of, both emotion and problem-focussed coping strategies, parents
have a combination of coping patterns identified by the Resiliency Model for managing their child’s
CF in the context of early surveillance. In fact, despite immediate distress and discomfort, it would
seem that early surveillance is a primary mechanism for coping with CF over the long-term.
Problem-focussed coping has been highlighted as an essential element that deserves more
attention for the purposes of implementing family resiliency practices for those families whose
children undergo early surveillance for CF lung disease. As families change and further coping
mechanisms are needed, problem-focussed coping is a promising avenue for assisting families to
develop coping strategies that currently do not receive much attention. Using the findings from my
research as a basis, promoting problem-focussed coping strategies could positively influence services
these families receive. Informing clinical practice through offering new ways to support families,
family resiliency practices can be developed and refined.
Thirdly, given the importance of appraisals in response to stress (Lazarus & Folkman, 1984)
and the importance of appraisals in adapting to paediatric chronic illness (McCubbin & McCubbin,
1993), developing a useful family schema is an essential process for families to engage in; because
the way in which a family defines a crisis has a critical impact on how it copes (Hawley, 2000). Family
201 Parenting Children with Cystic Fibrosis
schema describes the shared values, goals, priorities and expectations of family members, and is a
key element of highly resilient families (McCubbin & McCubbin, 1993). With knowledge that family
schema is beneficially influenced by early surveillance, clinicians and other HCPs working with these
families can help parents to construct or enhance their shared view of the world.
Family sense of coherence is a closely related construct to resiliency, which refers to a
measure of the extent to which families feel confident that the outcomes of a situation will be
favourable for them (Hawley, 2000). Sense of coherence originated from Antonovsy’s (1987)
salutogenic notion to health promotion. That parents whose children undergo early surveillance for
CF lung disease have hope for favourable outcomes is likely to contribute to their sense of
coherence, further validating the importance of family schema in the context of early surveillance.
Therefore, therapy from a resilience perspective can focus on how a common view as a competent
unit can be accessed, developed, enhanced and maintained as families seek to constantly adapt to
their child’s progressive condition. Moreover, encouraging a family to develop a collective view as
survivors of distress is not only clinically useful in the present but can equip families for facing
adversity in the future.
Lastly, my research has gone some way towards enhancing our understanding of how coping
with similar events becomes more effective over time. Parental coping appeared to become more
effective as knowledge about CF (e.g., prognosis), and experience with its treatment regimen (i.e.,
normalisation within the family setting, early surveillance procedures and home treatment) were
obtained. Accumulation of both knowledge and experience facilitated creation of expectations,
therefore changing the appraisal of the event in subsequent encounters. That is, more effective
coping was facilitated by being able to manage subsequent encounters with the new knowledge and
previous experience, which changes the appraisal of those subsequent encounters by building
expectations into the appraisal. Therefore, parents of older children are likely to cope better than
parents of younger children because they will likely have more, or different, coping skills that they
have developed over time due to their experiences and accumulation of knowledge.
By understanding both the practical and experiential encounters of an event, parents can
develop expectations and subsequent mechanisms to cope with future related events. Expectations
contribute to parents’ succeeding encounters by providing a psychological framework to orient
themselves for what is likely to occur in the future, along with how they will react, and how they will
cope. This practical and experiential orientation contributes to their ability to cope, or manage more
effectively, with subsequent experiences. This information can be used to develop targeted activities
aimed at supporting parental coping with their child’s condition across the 7 years a child undergoes
early surveillance. For example, talking to other parents with older children in the AREST CF program
202 Parenting Children with Cystic Fibrosis
may help parents to access that sense of experience and expectation through listening to other
parents. In other words – talking about what it is like for parents who have been through it can help
their understanding and their ability to cope. Additionally, a walk through the paediatric unit at the
treating hospital, exposing parents to the procedures and environment might help parents to create
expectations, and to begin to understand what their experience may be like, which may further
contribute to their understanding and their ability to cope.
In review, my research substantially adds to a growing body of literature on the importance
of facilitating adaptation to paediatric disease, as well as how parental appraisals and coping
resources and abilities are important factors in the process to adaptation. Moreover, my research
has highlighted how early surveillance contributes to parents’ appraisals and coping resources and
abilities. Accordingly, my research indicates a need to further characterise adverse outcomes that
might hinder adaptation. However more importantly, my research highlights that families cope well,
and appear resilient to burden of CF, because of early surveillance. Therefore, utilising a family
resiliency approach to support families in achieving adaptation that is tailored to their unique needs
and experiences will offer a modern and innovative approach to managing these families.
7.5 Further considerations: Future program development and implementation, and mental health
care of parents and implications for their families
The Cystic Fibrosis Foundation and the European Cystic Fibrosis Society Guidelines
Committee on Mental Health highlighted the inadequate and variable nature of CF mental health
care across centres in Europe and the US (Abbott, et al., 2015). Additionally, a survey about
experience of care administered in the US concurred with inadequate mental health care for
patients with CF and their families (Homa, Sabadosa, Marrow & Marshall, 2015). The same
disconnect seems to exist across Australian CF centres. A consensus statement released by the
committee (Quittner, et al., 2015) recommended that CF teams provide sufficient education to help
parents to develop coping skills and disease management.
I suggest developing guidelines for application of the Resiliency Model of Stress, Adjustment
and Adaptation to address critical elements of parent and family functioning within the context of
the AREST CF program. I believe that such an approach will ensure clinicians and other HCPs develop
strategies for intervention based on a systematic diagnosis and evaluation of parent and family
functioning under stressful circumstances. To implement additional services for parents and their
families within the hospital setting, infrastructure and finance support is needed from the hospital.
Evidence-based guidelines may support CF teams in leveraging funds for mental health professionals
where this aspect of healthcare has traditionally been overlooked (Havermans & Staab, 2016). The
social worker and/or psychologist require these additional resources if they are to support families
203 Parenting Children with Cystic Fibrosis
from a resiliency perspective and educate them on how to effectively cope with early surveillance.
For the psychologist's role within the multidisciplinary CF team to be effective, Kerem and colleagues
(2005) recommended that the post should be no less than 50% of their working time.
The Cystic Fibrosis Foundation and the European Cystic Fibrosis Society Guidelines
Committee on Mental Health also recommended annual screening of anxiety and depression in
parent caregivers, and if needed, referral for treatment to primary care or mental health services
(Quittner, et al., 2015). Duff et al., (2016) evaluated the feasibility of these recommendations with
65 parents of young children with CF during outpatient appointments. They suggested that,
depending on the size of the CF clinic, an annual 3-6 month screening period could be attainable.
The following diagram is an algorithm set out by the committee for screening and treatment of
depression and anxiety for parents and caregivers:
Figure 13. Screening and treatment of depression and anxiety: Algorithm for parents/caregivers
Preventative education and routine screening for anxiety and depression will create an
opportunity for appropriate preventive and supportive interventions instead of reactive therapeutic
intervention. Given the findings of my research, screening for depression should include awareness
that chronic sorrow is a likely mental health issue for parents, especially those who experience
multiple events associated with their child’s condition. Going beyond the traditional deficit-model of
psychological care is a modern approach to preventive and educational care for paediatric patients
and their families. Therefore, assessment of both psychopathologic and resiliency factors would
adequately address the multi-dimensional nature of how families cope with their child’s chronic
condition. Moreover, addressing mental health issues systematically is likely to improve health
outcomes, quality of life and reduce healthcare utilisation (Burker, Sedway & Carone, 2004; Smith, et
al., 2010).
Early surveillance techniques are promising, and clinical trials are needed to test which ones
are the best markers of disease and indicative of prognosis. Such outcome measures can be used as
clinical endpoints so that psychological distress experienced by parents can be substantiated, by
showing that early surveillance does indeed prolong children’s’ lives. Lastly, but just as importantly,
parents’ mental health and their ability to cope effectively are known significant predictors of family
204 Parenting Children with Cystic Fibrosis
functioning (Koutra, Triliva, Roumeliotaki, Lionis & Vgontzas, 2015; Pakenham & Cox, 2012;
Robitschek & Kashubeck, 1999; Saunders, 1999), and both child physical and mental health (Sheidow,
Henry, Tolan & Strachan, 2013; Szyndler, et al., 2005; Turner-Cobb & Steptoe, 1998). Moreover,
family functioning is also a significant predictor of both child physical and mental health (Everhart, et
al., 2014; Patterson, et al., 1990; Patterson, et al., 1993; Walsh, 2003). Therefore, considering
parents’ mental health as a primary predictor of how the psychosocial environment of the child
influences their health outcomes will ensure the child and their family is supported in a systemic way,
just as the family is a systemic unit.
In summary, my research offers important feedback about; the lived experience of parenting
a child with CF in the context of early surveillance for lung disease, parents’ involvement in the early
surveillance program, and it invites consideration of the development and implementation of
current and future practice and policy designed as a holistic approach to paediatric CF care.
Specifically, despite short-term adverse parental reactions and responses to their child’s unpleasant
experience for which they feel ultimately responsible, the long-term benefits to the child and the
family far outweigh immediate discomfort and distress. Importantly, my research has identified
areas of concern and suggested areas for improvement to support parents as they embark on such a
unique path of modern paediatric CF care. Undervaluing the non-clinical aspects of early surveillance
participation for parents limits the possibilities for addressing the holistic needs of parents’
psychological well-being. As early surveillance is inextricably tied to the CF experience, non-clinical
aspects of CF care are of paramount importance to the maintenance of their child’s health outside of
the clinical setting.
7.6 Strengths and significance of the research
My research has been responsive to the need for obtaining contemporary parental
perspectives, understandings and constructions of early surveillance for CF lung disease from both
mothers and fathers. As unique as early surveillance for CF lung disease is, so too are parents’
experiences that extend beyond current knowledge of parenting a child with CF. This information is
timely given the likely uptake of the early surveillance framework for future therapeutic intervention
trials in young children nationally and internationally.
A substantial strength of my research methodology was that it allowed for extension of
current understandings of parenting a child with CF beyond local boundaries resulting in data
collected from two national sites. Deriving themes from data collected with parents in Western
Australia and Victoria made it possible to demonstrate the lack of site-specific influence of early
surveillance on the construction of meaning that parents formed about their early surveillance
experience. In particular, a salient finding was that both adverse and beneficial experiences and
205 Parenting Children with Cystic Fibrosis
outcomes asserted by parents in the Western Australian context were also found to have relevance
for parents in Victoria, highlighting the significance of the findings as well as the unique contribution
of my research.
A further strength of my research was the involvement of parents to provide information
about their experiences of parenting within the context of early surveillance for CF lung disease.
People are best positioned to report their understandings of, and meanings in, experience that they
encounter (Denzin & Lincoln, 2005, 2008; Gelo et al., 2008; Patton, 2002), and it was therefore
appropriate to utilise a qualitative methodology to provide parents the opportunity to express and
explain their experiences from within the personal context with which they occur (Chwalisz et al.,
2008; Corbin & Strauss, 2008; Creswell, 2007; Janesick, 2000). By consulting with parents about their
experiences, I was able to gain insight into processes and principles of meaning-making about early
surveillance. An additional benefit of my research methodology was that I was able to identify
factors that both mediated and influenced how parents constructed understanding about their
child’s early surveillance for CF lung disease, their child’s condition and their parental role.
Moreover, this type of consultation allowed collection of in-depth information about the unique
experiences of parents that perhaps would not have been identified through the quantification of
personal experiences, or relying on secondary sources of information (Creswell, 2007).
Consequently, any actions resulting from this shared information concerning parents’ lived
experiences of parenting a child with in the context of early surveillance are more likely to be
responsive to their needs.
7.7 Limitations of my research
The primary aim of qualitative research is to generate findings that are beneficial and
authentic within the context in which data was collected, which means that limitations exist as to
the extrapolation of findings to other populations. Therefore, future research could assess the
applicability of these findings amongst other population groups across different clinical contexts and
settings. Additionally, given that participation in my research was voluntary, it is possible that some
parents were representative of; a more motivated sample, parents who had a vested interest in the
early surveillance program, or parents who were confident in discussing their experiences.
Accordingly, future research with parents who are less confident in discussing their experiences
would provide a more comprehensive account of the lived experience of parenting a child with CF in
the context of early surveillance. Moreover, the amount of time between interview and annual early
surveillance review varied because early surveillance reviews are conducted throughout the year at
different times. Therefore, parents’ recollection of events, and their associated emotions and
cognitions could vary depending on time between review and interview.
206 Parenting Children with Cystic Fibrosis
Whilst not a limitation as such, as an explorative research design, it is impossible to state any
sense of causality about how relationships and processes function to create outcomes. Therefore,
undefined (that is, bi-directional or associative) relationships have been identified that warrant
further investigation to determine direction and strength of relationships. Moreover, determining
whether any significant relationships function as predictors of health resilience are important issues
for future research so that mediators and moderators of health and family resiliency can be better
understood. Accordingly, a longitudinal design investigating parenting a child within the context of
early surveillance for CF lung disease to see if attitudes, outcomes and understandings change over
time would add value to my research findings.
Lastly, the data collection method requires consideration in terms of how it may have
affected the quality of my data. For optimal study recruitment, as well for convenience of data
collection for parents, the limitation imposed by conducting one and two-parent interviews was
deemed less important than obtaining a representative sample of both mothers and fathers.
However, different methods for data collection may have influenced information discussed with me
by potentially hindering couples’ responses because of fear or concern about a partner’s response to
certain information that they may have expressed more openly and honestly if interviewed on their
own. Therefore it must be acknowledged that methodological limitations of collecting data in this
way may have compromised findings, such that data quality and content may have been different if
parents were interviewed alone.
7.8 Directions for future research
Findings from my research emphasise the importance of shifting from a purely biomedical
perspective when exploring the lived experience of parenting children with chronic conditions and
relationships that mediate the experience. This involves continuing to challenge the unidimensional
biomedical model in clinical care, as well as non-family systems assumptions, theories and
perspectives in parenting children with chronic conditions. Findings from my research support a
salutogenic family resiliency perspective, that is, a holistic perspective that acknowledges the power
of the family in nurturance and development of its members. Therefore, I suggest that such an
approach be considered when addressing issues associated with parenting children with chronic
conditions. For example, acknowledging through policy and practice that pathways to family
adaptation and resiliency are influenced by parents’ appraisal of their ability to cope with their
child’s chronic condition within the family context with which it occurs.
Whilst descriptions and explorative research on parents’ experiences encompasses a holistic
approach to a child’s clinical care, both prior research and my research suggest that understandings
are still dominated by biomedical understandings of health (i.e., treatment regimen, nutrition)
207 Parenting Children with Cystic Fibrosis
despite the lived experience emphasising the importance of psychological and systemic factors in
facilitating sense of personal control and parenting competence in conducting home treatment as
parents adapt to managing their child’s CF. Therefore, to effectively promote treatment adherence
and a more holistic health message to the paediatric community, critical consideration needs to be
given to how knowledge about the lived experience of a child’s clinical care is better translated into
clinical policy and practice, particularly the psychological and systemic aspects. Moreover, my
research findings support recent theoretical and empirical literature that advocates for the use of
salutogenic models of psychological care for people experiencing adverse events (e.g., Antonovsky,
1987; Seligman & Csikszentmihalyi, 2000) suggesting the continued necessity for future models of
care to incorporate a strengths-based, rather than purely a deficit-based, perspective to supporting
parents adapting to their child’s chronic condition.
Findings from my research similarly indicated that the concepts of power and perceived
control from knowledge about early disease progression would benefit from future research. I
suggest these concepts and their relationships with perceived parenting competence and parenting
practices such as treatment adherence be further delineated so that parents and children can be
further supported in adapting to CF in the context of early surveillance. Moreover, with evidence
emerging from the data that making social comparisons influenced how parents constructed
meaning and that using social support influenced how parents managed their experiences, my
research suggests that social connections provide a powerful medium to promote adaptive
perspectives on paediatric chronic disease. Promoting parental and family adaptation to paediatric
chronic disease would benefit from future research, investigating how social networks may be used
to generate and sustain adaptive perceptions and how social connections can be effectively used to
translate knowledge about paediatric chronic disease into the delivery and implementation of
clinical agendas, policies and services.
To influence positive change in the experience of parenting a child undergoing early
surveillance for CF lung disease, findings from my research revealed that adverse parental reactions
and responses to both CF diagnosis and the early surveillance medical procedures are existing issues
warranting additional attention. Coping with psychological adversity was a substantial finding of my
research, pervasive across the condition and its clinical care. Fear, anxiety, guilt, chronic sorrow,
cognitive dissonance and ambivalence recognised from my research as impacting upon parents’
psychological well-being warrants further investigation. What role these outcomes play in the
construction of meaning in; parenting, CF, early surveillance is of particular importance to address
these issues. So too are the constructions of parenting a child within the context of early surveillance
for CF lung disease important. Additionally, my findings suggest that there is benefit in examining
208 Parenting Children with Cystic Fibrosis
whether parents whose children undergo early surveillance for CF lung disease experience more
adverse outcomes as a result of more and/or earlier hospitalisations than parents whose children
who have less and/or later hospitalisations, or whether parents whose children undergo early
surveillance are more resilient for future burden of CF for the same reasons.
Lastly, efficacy of the Resiliency Model is best demonstrated in studies designed to measure
several critical variables with the intention of determining which variables, in what causal order, help
to explain variability in family adaptation. Therefore, path analyses and structural equation
modelling could be considered to assess variables identified in my study as important factors that
might contribute to family adaptation within the context of early surveillance for CF lung disease.
Moreover, how processes and pathways are common to other paediatric chronic conditions would
shed light on commonalities and differences across those conditions that may be amenable to
intervention and/or support within the wider context of families living with paediatric chronic illness.
To conclude, although beyond the scope of my research, an important consideration for
future research based on my findings would be to further explore the role that clinicians, other HCPs
and the hospital system more broadly plays on parental thinking, in promoting and influencing
parental decision-making about their child’s clinical care. This would assist in making visible those
processes that maintain misconceptions about medical research and clinical components of early
surveillance. Taking into account the overwhelming and hectic nature of CF diagnosis and multi-
disciplinary nature of CF clinical care, identifying when and how such perceptions are developed and
maintained could lead to better clinical understandings on both clinicians’ and parents’ behalves.
7.9 Final considerations
A primary goal of my research was to address a gap in knowledge about the lived experience
of parenting a child undergoing a unique clinical care regimen for paediatric CF through consultation
with parents about their experience. A related goal was to highlight how this knowledge can be used
to develop policy and practice that more closely reflects parents’ lived experience of CF in the
context of early surveillance. In particular, this research aimed to understand influences on
constructed experience and attributions of meaning in clinical care with a particular interest in the
disjunction between policy and practice constructions and lived experiences. Although my research
has identified the importance of familial and psychological factors to parents’ experiences of their
child’s clinical care, findings suggest that parents’ understandings of early surveillance were
influenced by dominant social and biomedical discourses. It is important that these findings are used
to inform existing and future policy decision making and models of best practice towards promoting
adaptation and family resiliency for future burden of paediatric chronic disease. However, if this
209 Parenting Children with Cystic Fibrosis
knowledge is to be translated into practice so as to more closely align with parents’ lived
experiences, a critical review on how to more effectively translate policy into practice is required.
In conclusion, findings from my research encourage further thinking about how parents and
their families strive to achieve normality when their child is diagnosed with a chronic condition, and
more specifically, how fear, control and hope are implicated with knowledge of covert disease
progression in both practical and experiential terms. Furthermore, findings from my research also
provide evidence that parenting a child with a chronic condition is socially constructed, and therefore
a modifiable experience. With evidence that clinicians, other HCPs and dominant discourses have an
influence on parents’ thinking, positive aspects of early surveillance through parents’ experiences,
particularly the psychological aspects need to be more effectively communicated and promoted in
future clinical policy and research. Moreover, these aspects need to be reviewed as to how they can
be more effectively translated into practice, specifically that constructions of CF and parenting are
influenced by participation in an early surveillance program. Shifting the focus in this direction has
the potential to significantly modify parents’ constructions and understandings of parenting children
within the context of early surveillance, which could therefore influence the broader parenting and
family experience more positively, and ultimately influence children’s long-term health outcomes.
Lastly, I would like to reiterate the centrality of hope expressed by parents in my research
and the importance of hope for resilience and coping with adversities. The magnitude of hope, and
the effects it has in the daily lives of individuals, can be seen in President Barack Obama’s victory
speech following his campaign for the parliamentary leadership in 2008 that included the iconic
hope poster. His speech embraced and pulled at the heart-strings of the United States public. His
speech was one of hope; hope of a nation, and hope for a nation. His speech eloquently articulated
10 references of hope for the future, one of which was:
I’m not talking about blind optimism, the kind of hope that just ignores the enormity of
the tasks ahead or the road blocks that stand in our path. I’m not talking about the
wishful idealism that allows us to just sit on the sidelines or shirk from a fight. I have
always believed that hope is that stubborn thing inside us that insists, despite all the
evidence to the contrary, that something better awaits us so long as we have the
courage to keep reaching, to keep working, to keep fighting (Obama, 2008).
Obama’s expression of hope in this particular excerpt epitomises the hope expressed by
parents who took part in my research because; they do not underestimate the magnitude of their
situation, they are not simply observers of their situation but are proactive and action-oriented, and
despite the enormity of their situation, they are empowered to strive for a normal family life that
requires drastic and challenging changes to their initial expectations, roles and functions.
210 Parenting Children with Cystic Fibrosis
My view is that the salutogenic approach of parental support is a well-suited approach to
building and/or maintaining hope as a family characteristic that contributes to adaptation to
paediatric chronic disease and promotes family resilience for future burden – both normative and
disease-specific. The inherent link described at the beginning of my thesis between hope and
empowerment emanates from belief in choice, and belief that chosen actions can generate a future
different from the present (Groopman, 2005). Such beliefs cultivate motivation and capacity to
move forward despite present challenges. Early surveillance for CF lung disease is a catalyst for
empowerment through knowledge and a choice to act proactively, and belief that knowledge and
actions will bring about a future for children and families that is different from what has been
presented to them. Knowledge is constructed as control and the meaning attributed to control is
hope; hope for their child, hope for CF generations, hope for the future, and hope for a cure. Hope is
empowerment; motivation and capacity to strive for needs, desires and goals that might otherwise
seem insurmountable. Hope is coping with challenges and burdens. Hope is resilience. As such, my
thesis is a message of hope and resilience.
7.10 Postscript reflection
Notwithstanding ethical issues and challenges of investigating personal experiences of
parents who have chronically ill children, the emotional challenges I might experience as a
researcher was an aspect of humanistic research I failed to consider. As I read literature early in my
doctoral research, I sensed parents’ loss and sadness and I sensed their determination. My early
readings primed me to think about ethical relationships and meaningful research methodologies for
parents of chronically ill children. However, what it did not prepare me for was how to manage my
own personal responses and affective states while conducting my research. In varying degrees and
frequency, emotions and feelings such as sadness, empathy, enlightenment, passion, inspiration,
gratitude and joy accompanied my journey. Such responses underpinned questions about potential
biases I might hold as a researcher, as well as the place of qualitative psychological research in
exploring understudied phenomena in understudied populations within understudied contexts.
However, key to confronting and constructively analysing these experiences and thoughts
through the interactive researcher-participant dialogue (Ponterotto, 2005) was the reiterative
practice of deep critical reflection (Charmaz & McMullen, 2011; Schwandt, 2000). Such endeavours
were pivotal to challenging familiar assumptions of parenting practices, as well as opening my
naivety to the complexity of parenting children with chronic conditions. Lastly, it is important to not
overlook the ability of such parents to inspire and shape research. Throughout this journey I have
never stopped marvelling at parents’ capacity and commitment to their children and their families,
and at their resilience in the face of adversity. The daily treatment regimen parents must follow
211 Parenting Children with Cystic Fibrosis
alone is profound. Such commitment deserves respect. It is this role modelling that has equally
guided my own personal commitment as a researcher to the biopsychosocial model of clinical care
from a salutogenic approach.
212 Parenting Children with Cystic Fibrosis
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Appendix A
Invitational Recruitment Letter – Princess Margaret Hospital
Date Name
Address
Dear Parent/s I am writing to ask if you would be willing to be contacted by a researcher from The Telethon Kids
Institute who is undertaking a project called ‘The AREST-CF Early Surveillance Program for infants
and children with cystic fibrosis (CF): What do parents experience and how do they cope?’ and
wishes to recruit suitable parents from this Hospital. Parents from this Hospital are needed for this
study and from our records you would appear to be a potential participant.
Expected benefits to the participant and community
Potential benefits of taking part in this study to participants include:
Participants may experience the satisfaction of participating in research that may
benefit themselves, future patients and families
Participants will be able to be involved as consumer advocates for people with CF, and
their families
Participants may benefit from the reflection that follows answering the types of
questions and methods used in this study
Potential benefits of this study to the national CF community:
May enhance school and socio-emotional experiences and development within the CF community (nationally and internationally)
May lead to a larger study that will generate support for all children living with cystic fibrosis, which may improve school and socio-emotional outcomes
The aim of this study is to explore parents’ perspectives of what it is like to parent children who
undergo early surveillance of CF. We want to help parents by supporting them and their families.
This is very important as it may mean we can improve mental health and well-being for children
with CF and families by supporting them in the early years of children’s lives.
280 Parenting Children with Cystic Fibrosis
The people the researchers are looking for to help them with this study need to be:
The parents or guardians (primary carers) of children aged up to years with a documented diagnosis of CF (positive sweat chloride >60 mEq/L) and/or a genotype with two identifiable mutations consistent with CF accompanied by one or more clinical features with the CF phenotype; (“parents” in this context means any person/s defined by the family itself as the primary caregivers for the child/ren)
Parents of children with CF undergoing early surveillance with AREST CF
If you do not wish to hear more about this study, or be contacted further, please complete the
attached slip and return it in the envelope provided. If we do not hear from you within 3 weeks we
will assume you are willing to be contacted and the researcher, Cindy Branch-Smith, will contact
you shortly after to give you more information regarding the study. Should you wish to have
further information about the study before making a decision as to whether or not you wish to be
contacted please telephone the researcher (Cindy Branch-Smith on 08 94897601).
Whether or not you participate in this project, any future care you or your child receives at
this Hospital will not be affected in any way.
This study has been approved by the Princess Margaret Hospitals Ethics Committee, Edith
Cowan University Ethics Committee, Royal Children’s Hospital Human Research Ethics Committee,
and the confidentiality of all participants is assured.
Thank you for considering this
request. Yours sincerely
Doctor Tonia Douglas CF Centre Director
281 Parenting Children with Cystic Fibrosis
Appendix B
Information Statement and Consent Form – Princess Margaret Hospital
Information statement
The AREST-CF Early Surveillance Program for infants and
children with cystic fibrosis: What do parents experience and how do they cope?
Why are we doing the study?
Having a child with cystic fibrosis (CF) can be stressful for families and involve a lot of changes to lifestyle and family life. It can affect the health of family members too, especially emotional and mental health. Often the sicker the child is, the worse the impact of CF may be for families.
What we don’t know is whether how a family works, the emotional health of parents, their parenting style and the social circumstances of families affects how parents experience the early surveillance program or how families cope with their child being involved with the early surveillance program. In other words, do certain factors in the social and family background lead to healthy or unhealthy coping styles and strategies and do certain factors in the social and family background have an effect on how parents experience the early surveillance program?
This is very important as it will mean we can promote mental health and well-being of parents and children by better supporting families and helping them make changes to the family setting that promote better health.
The aim of this study is explore and understand caregivers’ coping and other experiences with being involved in the early surveillance program, and to understand if coping styles and strategies, and other experiences, are associated with specific domains within family and social systems.
Who is carrying out the study?
This study is being conducted by The Australian Respiratory Early Surveillance Team for Cystic Fibrosis (www.arestcf.org) at Princess Margaret Hospital for Children in Perth.
Do you have to take part?
No, your participation is purely voluntary, and if you choose not to take part that’s just fine and you will not experience any consequences of your decision. Your decision to participate will not affect your child’s care in any way. You may also choose to withdraw your consent at any time during the study.
What will you be asked to do if you decide to take part in this study?
Your child will need to do nothing extra if you participate in this program.
282 Parenting Children with Cystic Fibrosis
We will ask the parents to participate in an interview lasting between 1 and 1 ½ hours in length. During the interview, parents will be asked about their experiences with each of the clinical aspects of the program, including relinquishing your child for anaesthesia and obtaining annual results. Particular coping strategies that parents employ for each aspect of the program will also be established.
The interview will be carried out in hospital, at your home or another convenient place and we will make sure this happens at a convenient time for you if you choose to participate.
Is there likely to be a benefit to my child or family?
There may be no direct benefit to your child or your family by participating in this study, however, it is our experience that parents find thinking about their family, their children and how they parent quite a positive experience.
Is there likely to be a benefit to other people in the future?
This study looks at important questions for patients with CF and their families that we have yet to answer very well and will improve our understanding of the role of family and social factors in how families cope with being involved the program. This will help us to provide better all-round care for patients and families in the future.
What are the possible risks and/or side effects?
There are no anticipated side effects by taking part in this study.
What are the possible discomforts and/or inconveniences?
You may experience some inconvenience related to the time spent answering the interview questions as described above.
Where is your information kept?
The information we collect will be kept here at the hospital in a locked filing cabinet, held in a locked office. Information will be entered into a database that is password protected and accessed by the research team only.
What about my privacy?
The information we collect will be recorded in a separate file and kept strictly private. The study researchers will not know your name, as each parent will be given a codename. This further protects your family’s privacy.
Who has approved the study?
This study has been approved by the Ethics Committees at Princess Margaret Hospital, Edith Cowan University and James Cook University (Queensland).
Who do I contact for more information about this study?
If you would like any more information about this study, please do not hesitate to contact one of the research team. They are very happy to answer your questions.
Name Title Contact
Cindy Branch-Smith Principal investigator 9489 7818
283 Parenting Children with Cystic Fibrosis
Professor Julie Ann Pooley Primary supervisor 6304 5591
Dr Tonia Douglas Co-supervisor 9340 8830
Who to contact if you have any concerns about the organisation or running of the study.
If you have any concerns or complaints regarding this study, you can contact the Director of Medical Services at PMH (Telephone No: (08) 9340 8222). Your concerns will be drawn to the attention of the Ethics Committee who is monitoring the study.
What to do next if you would like to take part in this research:
If you would like to take part in this research study, please read and sign the consent form provided.
THANK YOU FOR YOUR TIME
284 Parenting Children with Cystic Fibrosis
FORM OF CONSENT
PLEASE NOTE THAT PARTICIPATION IN RESEARCH STUDIES IS VOLUNTARY AND PARTICIPANTS CAN WITHDRAW AT ANY TIME WITH NO IMPACT ON CURRENT OR FUTURE CARE. I ................................................................................................................................. have read Given Names Surname the information explaining the study entitled The AREST-CF Early Surveillance Program for infants and children with cystic fibrosis: What do
parents experience and how do they cope?
I have read and understood the information given to me. Any questions I have asked have been answered to my satisfaction. I understand I may withdraw from the study at any stage and withdrawal will not interfere with routine care for my child. I agree that research data gathered from the results of this study may be published, provided that names are not used. Dated ................................. day of ............................................................ 20 .......... Signature .................................................... I, ........................................................................... have explained the above to the (Investigator’s full name) signatory who stated that he/she understood the same. Signature .............................................................................................
285 Parenting Children with Cystic Fibrosis
Appendix C
Information Statement and Consent Form – Royal Children’s Hospital
The AREST-CF Early Surveillance Program for infants and children with cystic fibrosis: What do
parents experience and how do they cope?
Why are we doing the study?
Having a child with cystic fibrosis (CF) can be stressful for families and involve a lot of changes to
lifestyle and family life. It can affect the health of family members too, especially emotional and
mental health. Often the sicker the child is, the worse the impact of CF may be for families.
What we don’t know is whether how a family works, the emotional health of parents, their
parenting style and the social circumstances of families affects how parents experience the early
surveillance program or how families cope with their child being involved with the early surveillance
program. In other words, do certain factors in the social and family background lead to healthy or
unhealthy coping styles and strategies and do certain factors in the social and family background
have an effect on how parents experience the early surveillance program?
This is very important as it will mean we can promote mental health and well-being of parents and
children by better supporting families and helping them make changes to the family setting that
promote better health.
The aim of this study is explore and understand caregivers’ coping and other experiences with being
involved in the early surveillance program, and to understand if coping styles and strategies, and
other experiences, are associated with specific domains within family and social systems.
Who is carrying out the study?
This study is being conducted by The Australian Respiratory Early Surveillance Team for Cystic
Fibrosis (www.arestcf.org) at The Royal Children’s Hospital and Princess Margaret Hospital for
Children in Perth.
286 Parenting Children with Cystic Fibrosis
Do you have to take part?
No, your participation is purely voluntary, and if you choose not to take part that’s just fine and you
will not experience any consequences of your decision. Your decision to participate will not affect
your child’s care in any way. You may also choose to withdraw your consent at any time during the
study.
What will you be asked to do if you decide to take part in this study?
Your child will need to do nothing extra if you participate in this study.
We will ask you to participate in an interview lasting approximately 1 hour in length. During the
interview, you will be asked about your experiences with each of the clinical aspects of the program,
including relinquishing your child for anaesthesia and obtaining annual results. Particular coping
strategies that parents employ for each aspect of the program will also be established.
The interview can be carried out in hospital when you are visiting or staying, at your home or
another convenient place, or by TeleHealth (telephone video conferencing using secure internet
connection) or by telephone. We will make sure this happens at a convenient time for you if you
choose to participate.
If you do not wish to participate, please complete the opt-out form included with this letter and
return in the reply-paid envelope within 10 days. If you do not return the form, the principal
investigator will contact you about your interest in the study.
Is there likely to be a benefit to my child or family?
There may be no direct benefit to your child or your family by participating in this study, however, it
is our experience that parents find thinking about their family, their children and how they parent
quite a positive experience.
Is there likely to be a benefit to other people in the future?
This study looks at important questions for patients with CF and their families that we have yet to
answer very well and will improve our understanding of the role of family and social factors in how
families cope with being involved the program. This will help us to provide better all-round care for
patients and families in the future.
What are the possible discomforts and/or inconveniences?
You may experience some inconvenience related to the time spent answering the interview
questions as described above. If you experience any level of distress as a result of discussing your
experiences, you can be referred to the CF psychologist or CF clinic co-ordinator at the Royal
Children’s Hospital or an independent counselling service.
What are the possible risks and/or side effects?
There are no anticipated side effects by taking part in this study.
287 Parenting Children with Cystic Fibrosis
Where is your information kept?
The interviews will be audio-recorded and then destroyed once they have been typed into a Word
document. The documents will be assigned a code so that you cannot be identified. A copy of the
transcribed interview will be kept at RCH and a copy sent to the Telethon Institute for Child Health
Research in Perth where the principal investigator will complete the analysis of the interviews. The
documents at both sites will kept in a locked filing cabinet, held in a locked office. Information will
be entered into a Web application called SharePoint that is password protected and accessed by the
research team only.
What about my privacy?
The information we collect will be recorded in a separate file and kept strictly private. The study
researchers will not know your name, as each parent will be given a codename. This further protects
your family’s privacy.
Who has approved the study?
This study is a joint research project between the Royal Children’s Hospital and Princess Margaret
Hospital in Perth and has been approved by their respective Human Research Ethics Committees.
This study is a PhD collaboration at Edith Cowan University (Perth) and James Cook University
(Queensland) and has also been approved by their respective Human Research Ethics Committees.
Who do I contact for more information about this study?
If you would like any more information about this study, please do not hesitate to contact one of the
research team. They are very happy to answer your questions.
Name Title Contact
Cindy Branch-Smith Principal investigator 0416 580 255
Associate Professor John Massie Associate Investigator (03) 9345 5818
Judith Glazner Associate Investigator (03) 9345 5840
Who to contact if you have any concerns about the organisation or running of the study.
If you have any concerns and/or complaints about the project, the way it is being conducted or your
rights as a research participant, and would like to speak to someone independent of the project,
please contact:
Director, Research Development & Ethics, The Royal Children’s Hospital Melbourne on telephone:
(03) 9345 5044.
288 Parenting Children with Cystic Fibrosis
What to do next if you would like to take part in this research:
If you would like to take part in this research study, you don’t need to do anything now. A member
from the project team will contact you by phone to organise a convenient interview time and
location, and to answer any questions you may have. The consent form on the next page will be
collected at the meeting time prior to the start of the interview.
THANK YOU FOR YOUR TIME
289 Parenting Children with Cystic Fibrosis
CONSENT FORM
Research Project Title:
The AREST-CF Early Surveillance Program for infants and children
with cystic fibrosis: What do parents experience and how do they
cope?
Version Number: 1 Version Date: 01/08/13
I have read, or had read to me in my first language, the information statement version listed above and I understand its contents.
I believe I understand the purpose, extent and possible risks of my involvement in this project.
I voluntarily consent to take part in this research project.
I have had an opportunity to ask questions and I am satisfied with the answers I have received.
I understand that this project has been approved by The Royal Children’s Hospital Melbourne Human Research Ethics Committee and will be carried out in line with the National Statement on Ethical Conduct in Human Research (2007).
I understand I will receive a copy of this Information Statement and Consent Form
Participant Name Participant Signature Date
Declaration by researcher: I have supplied an Information Letter and Consent Form to the participant
who has signed above, and believe that they understand the purpose, extent and possible risks of
their involvement in this project.
Research Team Member Name Research Team Member Signature Date
Note: All parties signing the Consent Form must date their own signature.
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Appendix D
Western Australian Counselling Services
Cystic Fibrosis Western Australia (08) 9346 7333
Lifeline 13 11 14
Family Helpline (08) 9223 1100
Parenting WA Helpline 1800 654 432
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Appendix E
Victorian Counselling Services
Cystic Fibrosis Victoria (03) 9686 1811
Lifeline 13 11 14
Lifeworks 1300 543 396
Parentline Victoria 13 22 89
292 Parenting Children with Cystic Fibrosis
Appendix F
Personal Reflections
Parents have not had a healthy child to begin with (he spent his first few months in hospital) but
perceive him to be very healthy currently. Maybe their perception is based on the fact that they
started with an unhealthy child. They do not perceive the program as positive at all, but as a
necessity to the longevity of their child’s life. As XXA is only 2, they’re still learning about the
program. These parents (not just the interviewees) must be so resilient, not only for their
experiences, but to talk so openly about them.
Mum and Dad were willing to share their experiences openly. They were happy with their AREST CF
involvement. Maybe, mainly because XXA has been so well. It did seem that that may be the case. I
have expressed my thoughts previously about how I think parents’ perceptions/experiences may be
influenced by the health of the child.
Both parents were very open about their experiences and perceptions. Dad seemed like he felt that
if he had a better ‘measure’ of XXA’s disease severity, he would ‘deal with it’ better. Mum got quite
emotional when discussing her views of the PMH staff, specifically the nurses. This was not a
negative way, Mum was very grateful of the PMH staff. I wonder why you would choose to have a CF
child when you find out of their condition in-utero. In this interview, I got the first sense of the
evolution of coping. I identified some themes that have previously been discussed by other parents,
that is, distraction and viewing the illness as not as bad as others. A new theme that came out was
‘denial.’
Parent came across as the most confident and comfortable that I have interviewed in relation to her
involvement in the program and in terms of her ability to cope with the procedures and processes.
Her eldest son has CF and she agrees that has assisted her to deal with XXA’s CF in a more effective
way. She expressed how calm she is with the program and mentioned her belief in God as a major
factor in her ability to stay calm.
Parent felt much more comfortable with her involvement in the program (i. e., her experience) over
time, mainly due to knowledge and understanding of the procedures and processes that help to
keep her child healthy. Healthy child and found out at 4 months old-cancer diagnosis analogy? Does
this have any influence over tone/frame of mind of family life?
293 Parenting Children with Cystic Fibrosis
Appendix G
Interview Schedule
Before we start, I would to thank you for your participation in the research project. Your comments
and participation are greatly appreciated.
The purpose of this interview is to understand what you think, from your perspective, about your
experiences with having a child with cystic fibrosis in the AREST-CF early surveillance program.
Can you tell me a story about caring for your child with CF?
Can you tell me a story about your child?
How did that make you feel?
Have there been similar experiences?
Have there been different experiences that made you feel the same way?
Can you tell me what it is like for you, and your family, to be involved with AREST CF?
What do you think it is like for your child to be involved with AREST CF?
Have you learnt anything about yourself from being involved with AREST CF?
If so, what have you learnt?
Is there anything you would like to add that you think I would like to know?
Examples of probes
What was that like for you?
Why do you think that was the case?
Can you give me an example/do you have further examples of this?
294 Parenting Children with Cystic Fibrosis
Appendix H
Initial Coding of Interview Transcripts
CODE
TRANSCRIPT
cognitive restructuring
when you see some of those kids that are so physically or mentally disturbed, CF seems quite mundane
I kind-of normalise it and think it’s the same, maybe not exactly the same for every family, but every family has issues and problems
parental support I think we’ve started to let more people in, we’re talking about now with other people
I talk to other parents that are there waiting with you, it helps you along I guess, to have someone to talk to. You’re not totally on your own, you don’t quite feel like you’re the only one doing it, there is support around
being practical I think trying to stay on top of everything. I made lists so that I knew I had everything. I guess trying to be practical about it and just making sure that it all goes as smoothly as I could possibly make it
I just basically tried to get as organised as I could so I just made a list, making sure that we had everything, making sure that I was doing everything
distraction I just try to shut it out a bit and try and focus on other things, stay busy I guess
We just got on with our usual routine, I guess we preoccupy ourselves with that
denial It was a case of, it's in the back of your head but you don't really focus on it. You just push it back so you don’t think about it too much
I guess we kind-of almost put it to the back of our minds so we don’t think about it, so we don’t have to deal with it
MERGING CODES
Adaptive Coping Strategies
Maladaptive Coping Strategies
cognitive restructuring distraction
parental support denial
being practical
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Appendix I
Thematic Mind-Map - Trust
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Appendix J
Thematic mind-map – Cognitive dissonance